A Novel Technique For Short Nose Correction

Brief Clinical Studies
The Journal of Craniofacial Surgery
The Age-Related Development

of Maxillary Sinus in Children
Muhammet Degermenci, PhD, Tolga Ertekin, PhD,
Harun Ulger, PhD, Niyazi Acer, PhD, and
Abdulhakim Coskun, MDy
Introduction: Paranasal sinuses are complex structures and show
individual variation. Providing normative values for paranasal sinus
size and their changes related to age could be helpful in evaluating
the presence of some diseases related to sinonasal region. The
purpose of the current study was to investigate the development of
maxillary sinuses and evaluate the volume changes according to age
and sex by using stereological and ellipsoidal formula methods after
that to compare these approaches with each other in children.
Materials and Methods: This retrospective volumetric computed
tomography (CT) study was carried out on 361 individuals
(180 females, 181 males) between 0 and 18 years old (10 females,
10 males in each group, only 14 age group includes 11 males) with
no signs of sinus pathology volumetric estimations determined on
CT images using point-counting approach of stereological methods
and ellipsoid formula by using morphometric data.
Results: Maxillary sinus volume measurements that were obtained
using 2 methods were increased with age in both sexes until 16 years
old. There was a significant correlation determined between
2 methods (ICC 0.8941.000 for right and 0.8620.999 for left
maxillary sinus measurements). According to the sex, the right and
left mean maxillary sinuses volumes were determined at
8.30 5.19 and 8.57 5.53 cm3 in male and at 7.60 4.57 and
7.99 4.73 cm3 in female by using ellipsoid formula respectively.
By the stereological method these values were 8.28 5.26,
8.44 5.35 cm3 and 7.64 4.55, 7.85 4.73 cm3 respectively.
There was no statistically significant difference between the volume
of maxillary sinuses with sex and side using both methods.
Conclusions: This study presents the basic data for studies relative to
the development of the maxillary sinus in children according to
2 methods. The current study demonstrated that the point-counting
method and ellipsoid formula are both effective in determining volume
estimation of maxillary sinuses and are well suited for CT studies.
Key Words: Computed tomography, maxillary sinus, stereology,
volume
he paranasal sinuses are complex air-filled anatomical structures with a significant interindividual variation.1 A detailed
knowledge of the variation in the development of the paranasal
sinuses is in a clinically relevant matter for pediatric patients.2
Genetic diseases, environmental conditions, and past infectious
From the Department of Anatomy; and yDepartment of Radiology,
University of Erciyes, Kayseri, Turkey.
Received May 14, 2015.
Accepted for publication September 28, 2015.
Address correspondence and reprint requests to Tolga Ertekin, PhD,
Associate Professor, Department of Anatomy, School of Medicine,
University of Erciyes, Kayseri 38039, Turkey;
E-mail: tolga.ertekin@yahoo.com.tr
The authors declare that there is no conflict of interest.
Copyright # 2015 by Mutaz B. Habal, MD
ISSN: 1049-2275
DOI: 10.1097/SCS.0000000000002304
Volume 27, Number 1, January 2016
may affect these variations.38 The ostiomeatal complex is frequently imaged due to the infectious diseases of the nasal cavity and
paranasal sinuses.9 Therefore, more information about the developmental anatomy of the paranasal sinuses is critical in performing
procedures such as an intraoperative and a preoperative evaluation
of functional endoscopic sinus surgery (FESS).3,10
The maxillary sinuses (MSs) develop from the mesodermal
structures arising from the first branchial arch and their developments begin in the third week of gestation, and continue through
early adulthood.11 The MS penetration into the maxilla bone starts
before the birth.12,13 These sinuses are located in the maxillary bone
and situated behind the orbit and they are the largest sinus in the
paranasal sinuses. Some mechanisms are proposed to explain the
phenomenon of paranasal cavity development: nasal airflow, brain
growth, and formation of facial structures.14
Anatomical and developmental descriptions of the maxillary
sinus may be of great clinical importance. The pneumatization of
childrens sinuses and the pattern of normal development may serve
as a reference for evaluating normal or abnormal development of
the maxillary sinus. Normal values and clinical indexes can be used
in diagnostic, preoperative evaluation and treatment planning. An
understanding of age-related changes in the dimensions and volume
of the normal maxillary sinus may help in the evaluation of radiographs and identification of sinus abnormalities.15 The volumetric
value is the key to establishing an objective definition of hypoplasia
or sinus atelectasis. Chronic maxillary atelectasis (CMA), also
known as silent sinus syndrome, computed tomography scan of
paranasal sinus shows a fully developed opacified sinus with inward
retraction of sinus walls into the lumen with associated loss of the
sinus volume.16 It is useful to consider normal values and clinical
indices in the diagnostic approach to sinus pathologies such as
sinusitis or sinus dysmorphism. Therefore, the estimation of maxillary sinus volume (MSV) is important for a surgeon in FESS.9,17
The use of computed tomography (CT) instead of plain radiography in the work-up of paranasal sinus imaging was recommended at the beginning of the 1990s.1 The introduction of CT with
axial, sagittal, and coronal section has allowed a more exact
assessment of these structures. Moreover, the application of morphometric procedures using an ellipsoid formula to these radiological images adds a new perspective volume estimation of MSs.18
The different anatomical dimensions of the paranasal sinuses can
also be obtained from CT images.19 The Cavalieri principle of
stereological approaches allows the researchers to obtain the
volume of the object of interest using sections or section images.
By using stereological methodologies, many numerical values such
as volume, surface area, and coefficient error can be found.4
The value of our study lies in the fact that a few studies in the
literature have investigated the MSV by using the stereological
method, and however, no study has made comparison between the
stereological and ellipsoidal formula methods. This study was
designed to fill this gap in knowledge, so we compared the
MSV using the stereological and ellipsoidal formula methods
and we determined MS development in children by both methods.
METHODS
This study was performed retrospectively on axial, sagittal, and
coronal scan images taken from 361 individuals (180 females and
181 males) aged between 0 and 18 years who had been admitted to
Erciyes University Medical Faculty. The individuals ages were
equally distributed with 20 individuals (10 females, 10 males, only
14 age group includes 11 males) in each age group. Subjects who
had symptoms of sinus pathology such as fracture, cysts of tumor
and persons with congenitally absent sinuses were excluded from
the study. The present study was approved by the ethical committee
of Erciyes University, Turkey.
# 2015 Mutaz B. Habal, MD

e38
Copyright 2015 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
Computed Tomography Procedure

We analyzed the intact cranial CT images of all the subjects. The
CT images were prepared using the following protocol. The axial
CT scans of cranial images were obtained using a CT scanner
(Multislice 16 detector GE) applying the following parameters: kV,
120, mAs, 130, FOV (field of view), 2425 cm, section time, 2.7 s,
slice thickness, 1.3 mm. CT images were taken from an axial plane
from picture archive and communication system (PACS) and then
converted into coronal and sagittal planes with 0.7-mm thickness
using DicomWorks software.
FIGURE 2. Estimation of MSV on coronal CT images by superimposing

randomly the point-counting grid.
Volume Estimation
We used 2 different techniques for calculation of the MSs:
1. Ellipsoid formula using morphological parameters.
2. Stereological analysis using the Cavalieri principle (pointcounting method).
Ellipsoid Formula
In each subject the largest diameter was evaluated on 3 axes. The
maximal extension in the 3 plans was defined as the highest
dimension noted for the length on sagittal images, and height as
maximal extension on coronal images. The width used for this
calculation was the mean value of the maximal width and the width
at the middle of the MS on the axial images (Fig. 1). The MSV was
calculated by the simplified ellipsoid volume formula using these
morphometric data (Eq. 1).
MSV maximal width maximal height maximal depth
0.520,21
(1)
The Cavalieri Principle Applied to Computed

Tomography Sections and Stereological Analysis
The point-counting method is based on the Cavalieri principle.
The Cavalieri principle has been used for volume estimations in the
literature.22,23 Using the Cavalieri method (point-counting), an estimate of the volume of a structure of irregular shape and size may be
obtained influentially and with known precision.24 According to this
method, the CT images of a section series of 1.3-mm thickness were
used to estimate the structures volume. The films were saved on a
computer and the transparent square grid test system with d 0.3 cm
between test points was superimposed, randomly covering the entire
image frame (Fig. 2). The points that hit the structure sectioned
surface area were counted for each section and the MSV was
estimated using the modified formula (Eq. 2).9,25,26

SU d 2 X
V t

P
(2)
SL
where t is the section thickness of consecutive sections, SU is
the scale unit of the printed film, d is the distance between the points
of the grid, SL is the measured length of the scale printed on the film.
SP is the total number of points that hit the sectioned cut surface
areas of MS.4,5,27
FIGURE 1. Measurements for ellipsoid formula (maximal width (B), maximal

width at the middle (A), maximal height (C), maximal length (D)).
2015 Mutaz B. Habal, MD
Coefficient of Error for Point Counting

The coefficient of error (CE) of the point-counting method was
calculated using the formula described in previous studies.23 A CE
value lower than 5% is an acceptable range according to the
literature.25 It is important to note that the CE has no real biological
meaning. Rather, it is most useful for evaluating the precision of
stereological estimates. It is also important to note that an appropriate grid size and the number of slices required for volume
estimation of an object are crucial at the beginning.28
Statistical Analysis
The comparison of the volume results between the sexes was
analyzed using the independent t test and the comparison of the
volume results between right and left MSs was analyzed using
the paired sample t test. The estimation of the effects of aging on the
size of the MSs was applied using a Pearson correlation test. Results
have been expressed as the number of observations and mean
standard deviation (SD). A P value less than 0.05 was considered
statistically significant. All statistical analyses were performed with
the Statistical Package for the Social Sciences software (SPSS Inc,
Chicago, IL).
RESULTS
Estimation of Maxillary Sinus Volume Using
Ellipsoid Formula
To get MSV morphometric measurements were taken. The
morphometric results showed that upper and lower values of height,
depth, and width of MS were 0.53 to 5.01 cm (mean 2.60 0.82),
0.53 to 4.16 cm (mean 2.87 0.66), and 0.32 to 3.09 cm
(mean 1.88 0.48) respectively. The mean values of height,
depth, and width were estimated at 2.63 0.84, 2.92 0.67,
and 1.89 0.47 cm for male, 2.82 0.66, 2.81 0.66, and
1.86 0.49 cm for female respectively. The average values of
width, height, and depth measurements of bilateral MSs according
to age are given in Table 1.
Using the ellipsoid formula, the mean MSV was calculated as
8.11 5.02 cm3. The mean volumes of right and left MSs were
7.95 4.90 and 8.28 5.15 cm3, respectively. There was no statistically significant difference between right and left MSs in each
age group separately. There was a positive correlation between age
and MSV (Table 2).
According to the sex, for men, the right and left mean MSV were
determined at 8.30 5.19 and 8.57 5.53 cm3, for women, these
values were calculated as 7.60 4.57 and 7.99 4.73 cm3 respectively. While statistical differences were not determined bilaterally
at any age group in men and were only determined at 5 years of age
group in women (P < 0.05) (Table 3). The comparison between
female and male volume measurements revealed no significant
differences and MSV reached a maximal size at 16 years of age
(Fig. 3).
e39
TABLE 1. Mean Values of Maximal Width, Height, and Depth of MS

Width (cm)
Height (cm)
Depth (cm)
Age
Right
Left
Right
Left
Right
Left
1
2
3
4
5
6
7
8
9
10
11
12
13
14
15
16
17
18
20
20
20
20
20
20
20
20
20
20
20
20
20
21
20
20
20
20
0.84
1.22
1.46
1.68
1.70
1.77
1.85
2.05
2.06
2.04
2.03
2.01
1.89
1.99
2.17
2.12
2.20
2.14
0.88
1.20
1.45
1.74
1.65
1.93
1.92
2.08
2.03
2.09
2.10
2.19
2.01
2.12
2.24
2.33
2.31
2.21
1.10
1.43
1.87
2.05
2.18
2.25
2.47
2.45
2.48
2.63
2.81
3.05
3.02
3.20
3.44
3.55
3.42
3.35
1.11
1.43
1.90
1.98
2.21
2.23
2.47
2.49
2.53
2.59
2.80
2.96
3.10
3.18
3.47
3.50
3.42
3.47
1.33
2.07
2.32
2.46
2.67
2.56
2.91
2.92
2.84
3.12
3.15
3.18
3.18
3.34
3.34
3.50
3.44
3.36
1.34
2.05
2.33
2.47
2.59
2.49
2.77
2.91
2.94
3.06
3.17
3.07
3.28
3.34
3.33
3.53
3.54
3.36
n, case number.
Estimation of Maxillary Sinus Volume Using

Stereological Method
The MSV that was obtained using the stereological method on
CT images showed no statistical difference between the 3 plans
(axial, coronal, and sagittal) (Table 4; Figs. 4 and 5). The coefficient
of error (CE) was determined on each 3 plans; therefore the
accuracy of measurement was calculated. The mean CE values
were determined as 2.41 to 2.39 on a coronal plan, 2.35 to 2.34 on a
TABLE 2. Mean Volumes of Bilateral MSs Calculated With Ellipsoid Formula by
Using Morphometric Data
Age
1
2
3
4
5
6
7
8
9
10
11
12
13
14
15
16
17
18
n
20
20
20
20
20
20
20
20
20
20
20
20
20
21
20
20
20
20
Right MSV (cm3)
Left MSV (cm3)
Mean SD
Mean SD
0.95 1.82
1.81 0.92
3.26 1.22
4.34 1.70
5.01 1.46
5.29 1.86
6.68 1.28
7.60 2.71
7.52 2.84
8.66 2.67
9.26 3.11
10.11 3.08
9.61 3.08
11.32 4.85
12.70 3.23
13.61 4.99
12.48 4.98
12.74 5.50
0.98 1.68
1.81 0.88
3.32 1.22
4.42 2.14
4.78 1.50
5.51 1.84
6.61 1.58
7.75 2.56
7.79 2.70
8.48 2.58
9.52 3.38
10.19 2.85
10.36 2.08
11.97 5.08
12.71 4.25
14.81 4.85
14.27 3.65
13.54 5.88
MSV, maxillary sinus volume; n, case number.
e40
r
0.992
0.876
0.977
0.932
0.850
0.928
0.800
0.838
0.898
0.900
0.940
0.963
0.838
0.891
0.705
0.850
0.614
0.919
sagittal plan, and 3.34 to 3.32 on an axial plane for right and left
MSV, respectively. In this study, the CE means in both age and plan
were determined to be lower than 5%.
The mean stereological volume was calculated by averaging
the measurements obtained from the 3 plans. The mean volumes
of right and left MSs were calculated as 7.96 4.92 and
8.15 4.99 cm3 respectively, and no significant statistical difference was determined on bilateral MS measurements in each age
group (Table 5). In addition, positive correlation was found between
age and MSV in stereological measurements and MSV reached a
maximal size at 16 years of age (Fig. 6).
According to the sex the mean volumes of the right and left MSs
were calculated as 8.28 5.26, 8.44 5.35 and 7.64 4.55,
7.85 4.73 cm3 for men and women respectively. When the bilateral values were compared, a significant statistical difference was
determined only 5 years age group in females (P < 0.05) but no
difference in males (Table 6). In addition, when the MSV calculated
using the stereological method compared with the sex, no statistical
difference was determined (Fig. 6).
Comparison of Stereological Method and

Ellipsoid Formula
In this study, volume measurement of maxillary sinus was made
by means of morphological parameters with the ellipsoid formula
and based on the Cavalieri principle using the stereological method.
When the measurements that were obtained by using 2 methods
were compared, a significant correlation was determined between
the results. Interclass correlation coefficient (ICC) values were
observed between 0.894 and 1.000 (mean 0.981) and between
0.862 and 0.999 (mean 0.978) for right and left maxillary sinus
measurements, respectively. These results showed that there was a
statistical compatibility between the ellipsoidal formula and the
Cavalieri principle method measurements. Average volume
measurements calculated by 2 methods for each age group are
given in Table 7. The agreements between methods were subjected
to BlandAltman plots using volume differences of 95. This
showed that the volumes estimated by the stereological (pointcounting) method and the ellipsoid formula for right MS differed by
between 1.3 and 1.3 cm3 (Fig. 7) and differed by between 1.4 and
1.2 cm3 for left MS (Fig. 8).
DISCUSSION
0.677
0.984
0.320
0.686
0.225
0.179
0.760
0.681
0.359
0.512
0.316
0.691
0.071
0.213
0.989
0.061
0.058
0.136
Despite the clinical importance of paranasal sinuses, the real

function of paranasal cavity has not yet been clarified. Regardless
of its function, the fact that the paranasal sinus constitutes the major
cavity of human skull indicates that its most important anatomical
feature is the air space of the cavity itself.4,19 Paranasal sinus
anatomy is complex and shows individual differences. Significant
differences in structure between the 2 sides may also exist in the
same person.2931
Providing normative values for paranasal sinus size and their
changes related to age could be helpful in evaluating the presence of
any abnormality. The volume measurements of structures and
organs in our body are important for diagnosis of some diseases.4,29,32 In volume estimation, the inaccurate results were
determined because of physical effects (etc., methods and imaging),
so there is a need for an easy, cheap, and highly reliable method to
calculate the volume of organs.33 The sinonasal region is frequently
imaged because of infectious diseases of the nasal cavity and
paranasal sinuses. The volumetric value is the key to establishing
an objective definition of sinus hypoplasia. In the current day,
knowledge of paranasal sinus anatomy is very important for FESS
and it is essential for an operational evaluation.9,18
#
TABLE 3. Bilateral MSV Results in Both Sex Using Ellipsoid Formula

Males
Right MSV (cm3)
Left MSV (cm3)
Age
Mean SD
Mean SD
1
2
3
4
5
6
7
8
9
10
11
12
13
14
15
16
17
18
1.41 2.53
1.75 0.45
3.52 1.50
4.65 2.13
4.91 1.66
5.81 1.81
6.60 1.14
7.53 2.94
7.50 2.88
8.73 2.60
9.53 1.31
9.91 3.75
10.52 4.00
11.44 3.79
13.63 3.10
14.27 6.35
13.73 5.17
13.54 6.74
1.44 2.31
1.73 0.54
3.54 1.52
4.73 2.73
4.70 1.81
6.16 1.49
6.29 1.70
7.11 2.53
7.71 2.70
8.33 2.36
9.51 2.02
9.84 3.43
11.14 2.43
11.24 4.37
13.60 5.11
16.21 6.00
14.67 3.92
14.39 7.08
Females
Right MSV (cm3)
Left MSV(cm3)
Mean SD
Mean SD
0.820
0.929
0.814
0.806
0.582
0.134
0.331
0.296
0.779
0.257
0.978
0.827
0.392
0.248
0.980
0.090
0.455
0.235
0.49 0.34
1.88 1.25
3.00 0.84
4.03 1.17
5.10 1.30
4.69 1.86
6.75 1.46
7.67 2.62
7.49 2.96
8.59 2.88
8.98 4.31
10.32 2.42
8.71 1.50
11.19 6.02
11.77 3.25
12.94 9.36
11.24 4.72
11.82 3.84
0.52 0.42
1.89 1.15
3.10 0.86
4.11 1.40
4.85 1.20
4.81 1.97
6.93 1.48
8.38 2.56
7.87 2.85
8.64 2.91
9.50 4.49
10.53 2.27
9.581.37
11.68 6.00
11.82 3.20
13.41 3.06
13.88 3.53
12.20 3.18
0.722
0.920
0.252
0.753

0.028
0.737
0.565
0.204
0.197
0.917
0.264
0.395
0.057
0.565
0.939
0.463
0.075
0.363
MSV, maxillary sinus volume.

P < 0.05.
There are many studies evaluating the volume of the paranasal

sinuses. Some researchers estimated the volume of paranasal sinuses
on dry skulls,34 cadaveric specimens,35 CT images,18 and MR
images9 using either only morphological or only stereological
methods. Except these 2 methods, the studies in which 3-dimensional
reshaping methods used with the help of computer programs can be
found easily.36 In our study, the MSV was measured by means of
morphological measurements using the ellipsoid formula and based
on the Cavalieri principle on CT images. There is no study in which
the MSV is calculated using both methods compared with each other.
Stereological methods provide some data to the researcher making
appropriate changes to the sampling procedures. Therefore, the
method applied here supplies a CE of measurements with each
volume, giving as a percentage the potential variability in any given
volume measurement. When the CE of these measurements is bigger
than 5%, it can generate obvious problems with accuracy and hence
interpretation. These problems may arise if too few slices or too few
points are to take into count. The mean CE estimate of the paranasal
sinuses in this study was less than 5%, meaning that the sectioning
interval or section number and the point density of the transparent grid
were sufficient to obtain reliable data.4,25
The different anatomical dimensions of the paranasal sinuses
can be obtained from CT images. These morphometric measurements were used for volume quantification of MSs by the ellipsoidal
formula in different studies.9,26 In literature, MSV were calculated by using the ellipsoid formula as 14.4 cm3 and 18.3 cm3 for
the subjects aged 18 to 33 years and aged 16 years, respectively.9,26 In our study, the MSV at 16 to 18 years was determined
as 14.20 cm3 with the same method. Despite the age difference,
this similarity is due to no changes of the MS as volumetric after
the age of 16.
FIGURE 3. The comparison of the MSV according to sex. MSV, maxillary sinus
volume.
A, axial plan, C, coronal plan, MSV, maxillary sinus volume, S: sagittal plan.
TABLE 4. Intraclass Correlation Coefficients (ICC) Among 3 Plans for Right and
Left Measurement
Right MSV ICC
Left MSV ICC
Age
C-S
C-A
S-A
C-S
C-A
S-A
1
2
3
4
5
6
7
8
9
10
11
12
13
14
15
16
17
18
20
20
20
20
20
20
20
20
20
20
20
20
20
21
20
20
20
20
0.997
0.986
0.994
0.981
0.944
0.914
0.848
0.965
0.923
0.971
0.997
0.988
0.967
0.996
0.966
0.983
0.973
0.994
0.998
0.976
0.990
0.961
0.904
0.947
0.776
0.914
0.957
0.950
0.995
0.981
0.989
0.998
0.963
0.975
0.953
0.996
0.998
0.984
0.991
0.955
0.929
0.911
0.864
0.949
0.926
0.958
0.996
0.974
0.975
0.998
0.944
0.957
0.960
0.999
0.979
0.986
0.979
0.950
0.923
0.959
0.988
0.978
0.942
0.979
0.997
0.981
0.941
0.999
0.939
0.986
0.972
0.999
0.995
0.990
0.966
0.929
0.896
0.952
0.987
0.924
0.951
0.975
0.996
0.970
0.940
1.000
0.960
0.958
0.948
0.999
0.991
0.989
0.973
0.960
0.962
0.934
0.986
0.942
0.904
0.957
0.997
0.972
0.929
1.000
0.970
0.979
0.933
1.000
e41
TABLE 5. Mean Volumes of Bilateral MSs Calculated by Using Stereological

Method
FIGURE 4. The comparison of volume results determined on 3 plans calculated

by using stereological method for right MS. MSV, maxillary sinus volume.
Right MSV (cm3)
Left MSV (cm3)
Age
Mean SD
Mean SD
1
2
3
4
5
6
7
8
9
10
11
12
13
14
15
16
17
18
20
20
20
20
20
20
20
20
20
20
20
20
20
21
20
20
20
20
0.95 1.60
1.77 0.85
3.20 1.26
4.33 1.71
4.98 1.49
5.22 1.88
6.60 1.36
7.59 2.75
7.46 2.84
8.08 3.06
9.35 2.89
9.87 2.84
9.60 3.00
11.48 4.78
12.56 2.90
13.99 4.78
13.19 4.66
12.95 5.43
0.91 1.45
1.78 0.76
3.28 1.20
4.32 1.96
4.72 1.56
5.46 1.85
6.61 1.62
7.78 2.77
7.77 2.66
8.07 3.08
9.34 3.19
9.96 2.74
10.31 2.00
11.80 4.84
12.71 2.98
14.74 4.24
13.86 3.80
13.05 5.56
0.993
0.857
0.970
0.917
0.857
0.939
0.816
0.851
0.887
0.942
0.974
0.922
0.822
0.909
0.759
0.873
0.789
0.952
0.548
0.924
0.259
0.947
0.158
0.121
0.969
0.569
0.308
0.936
0.948
0.727
0.087
0.480
0.748
0.166
0.307
0.805
For the MSV that was calculated by using the Cavalieri principle, Karakas et al29 reported a mean volume of 11.54 5.10
and11.82 5.38 cm3 in subjects between 5 and 55 years for right
and left sides respectively. Another study that used the subjects
between 18 and 72 years, the MSV was determined to be
18.0 6.0 cm.3,4 Similarly, in our study, these volume results were
found to be 7.96 4.92 cm3 for the right and 8.15 4.99 cm3 for the
left. Therefore, the differences in volume values were caused by
using different age groups. In addition, there is no study which
calculated MSV on 3 different plans by using the stereological
method. Studies that calculated MSV only used 1 plan for volume
estimation and so they did not compare and investigate their results
accuracy 4,29 In our study, the MSV was measured with the
stereological method, on CT images obtained by such 3 different
plans as axial, coronal, and sagittal. No statistically significant
difference was determined according to the volume measurements
obtained from each 3 plans and compatibility between results
was obtained.
Growth of paranasal sinuses starts as prenatal and they continue
growing toward each direction depending on enlargement of other
nasal bones and nasal cavity between the ages of 1 and 7. At birth,
the MS appears as a small sac; it has 2 pneumatizations, the first
between birth and 2 years of age and the second between 7 and
12 years of age, with slow development between 14 and 18 years of
age.32,37 The MS reach adult dimensions at 14 to 18 years of age or
at 15 years, and during the teenage years, according to various
reports.32,38 Similar to the other studies, in our study, the MSV
reached a maximal volume at 16 years of age and there were no
significant changes in the MSV after the age of 16. Studies reported
that MS increases regularly with age.29,38,39 Similarly, Schatz et al40
observed that MS exhibited an increase in volume for a period up to
15 years, and then maintained their size.
On the contrary, Emirzeoglu et al. reported that the MSV tends
to decrease after the age 20 years, likewise we reported that MSV
was slightly decreased after the age of 16 years in both sexes.
We thought that age differences among the studies may be derived
from the subjects race, sex, and methods that were used in
researches.19,41
In the literature, researchers reported that there was no statistically significant difference in the MSV between the left and right
sides when considering all ages.19,26,41 In addition, Barghouth et al9
reported that a leftright difference existed for the MS when the age
is over 8 years old and they found that the right MS length was
systematically greater than the left one. In the current study, there
were no differences in the MSV between the left and right sides in
each age group at either stereological method or ellipsoid formula.
We also estimated that the left MSV was greater than the left one in
all ages. Similarly, Nowak and Mehls42 reported that the MS of left
side were greater than the right side in both sexes.
Some authors have reported difference of the MS between males
and females,4,9,19 whereas others have shown no such difference.18,43 Barghouth et al9 found no statistically significant differences for growth curves for boys and girls in MSs. Nevertheless,
Spaeth et al2 found that most of the sinuses of female children are
significantly larger than those of boys of the same age until the age
of 5 or 6 years. Nowak and Mehls42 reported that the MSs were
greater in females than males. Some studies found that men had a
greater difference in MSV than women among the different age
groups.26,29,35 Our results showed that the MSV is larger in males,
but we found no significant statistical difference, according to sex in
all ages for both methods.
As a result, when comparing our results to the literature, we
thought that the differences among the studies are due to the number
of individuals used, age range handled, and methods used for
calculating the MSV. We have not, however, seen any study
FIGURE 5. The comparison of volume results determined on 3 plans calculated

by using stereological method for left MS. MSV, maxillary sinus volume.
FIGURE 6. The comparison of the MSV according to sex. MSV, maxillary sinus
volume.
e42
TABLE 6. Bilateral MSV Results in Both Sex Using Stereological Method

Males
Females
Right MSV (cm3)
Left MSV (cm3)
Age
Mean SD
Mean SD
1
2
3
4
5
6
7
8
9
10
11
12
13
14
15
16
17
18
1.34 2.22
1.75 0.47
3.56 1.55
4.61 2.18
4.94 1.69
5.79 1.79
6.56 1.34
7.46 2.97
7.49 2.85
7.82 3.47
9.70 1.41
9.65 3.59
10.51 3.80
11.61 4.22
13.39 3.00
14.67 6.19
13.90 5.29
14.03 6.82
1.29 1.99
1.78 0.62
3.54 1.50
4.55 2.45
4.76 1.83
6.13 1.49
6.33 1.77
7.06 2.56
7.69 2.59
7.66 3.51
9.50 1.87
9.45 3.35
11.08 2.42
12.02 4.13
13.88 2.96
16.07 5.32
14.47 4.38
14.30 7.04
Right MSV (cm3)
Left MSV (cm3)
Mean SD
Mean SD
0.617
0.850
0.852
0.829
0.613
0.133
0.461
0.338
0.711
0.491
0.495
0.428
0.431
0.473
0.507
0.175
0.596
0.670
0.55 0.39
1.79 1.14
2.85 0.81
4.06 1.14
5.03 1.36
4.66 1.81
6.63 1.45
7.71 2.67
7.44 2.98
8.35 2.75
9.00 3.93
10.09 1.99
8.68 1.65
11.33 5.56
11.74 2.69
13.32 2.97
12.49 4.09
11.88 3.62
0.54 0.42
1.78 0.91
3.03 0.82
4.09 1.41
4.67 1.35
4.79 2.01
6.88 1.50
8.50 2.19
7.85 2.87
8.48 2.72
9.17 4.23
10.46 2.00
9.55 1.13
11.56 5.75
11.54 2.63
13.42 2.43
13.25 3.25
11.80 3.48
0.769
0.905
0.062
0.885

0.035
0.544
0.411
0.142
0.192
0.769
0.392
0.410
0.077
0.767
0.746
0.800
0.358
0.867

P < 0.05.
evaluating the relationship between the stereological method and

the ellipsoid formula that was used for the volume estimation of the
MS. When volume values of MS that were measured by using both
methods bilaterally based on age are compared, a positive correlation between the 2 methods was observed. In fact, the current
study demonstrated that the point-counting method and the ellipsoid
formula are both effective in determining volume estimation of MSs
TABLE 7. Comparison of the 2 Methods for Both Sides
Stm.
Right (cm3)
Ellipsoid
Right (cm3)
Stm.
Left (cm3)
Ellipsoid
Left (cm3)
Age
Mean SD
Mean SD
ICC
Mean SD
Mean SD
ICC
20
0.95 1.60
0.95 1.82
0.995
0.91 1.45
0.98 1.68
0.994
20
1.77 0.85
1.81 0.92
0.984
1.78 0.76
1.81 0.88
0.968
20
3.20 1.26
3.26 1.22
0.993
3.28 1.20
3.32 1.22
0.995
20
4.33 1.71
4.34 1.70
0.998
4.32 1.96
4.42 2.14
0.995
20
4.98 1.49
5.01 1.46
0.995
4.72 1.56
4.78 1.50
0.990
20
5.22 1.88
5.29 1.86
0.999
5.46 1.85
5.51 1.84
0.999
20
6.60 1.36
6.68 1.28
0.993
6.61 1.62
6.61 1.58
0.996
20
7.59 2.75
7.60 2.71
0.996
7.78 2.77
7.75 2.56
0.994
20
7.46 2.84
7.52 2.84
1.000
7.77 2.66
7.79 2.70
0.999
10
20
8.08 3.06
8.66 2.67
0.915
8.07 3.08
8.48 2.58
0.943
11
20
9.35 2.89
9.26 3.11
0.992
9.34 3.19
9.52 3.38
0.992
12
20
9.87 2.84
10.11 3.08
0.990
9.96 2.74
10.19 2.85
0.984
13
20
9.60 3.00
9.61 3.08
0.993
10.31 2.00
10.36 2.08
0.991
14
21
11.48 4.78
11.32 4.85
0.977
11.80 4.84
11.97 5.08
0.984
15
20
12.56 2.90
12.70 3.23
0.976
12.71 2.98
12.71 4.25
0.862
16
20
13.99 4.78
13.61 4.99
0.981
14.74 4.24
14.81 4.85
0.971
17
20
13.19 4.66
12.48 4.98
0.894
13.86 3.80
14.27 3.65
0.968
18
20
12.95 5.43
12.74 5.50
0.991
13.05 5.56
13.54 5.88
0.993
361
7.96 4.92
7.95 4.90
0.989
8.15 4.99
8.28 5.15
0.991
Total
ICC, interclass correlation coefficient; Stm, stereological method.
and are well suited for CT studies. The stereological technique may
provide quick, unbiased, and reproducible estimations. Any significant difference and the good agreement between the pointcounting method and the ellipsoid formula give strong evidence
about the reliability of the 2 methods. For maxillary sinus volume,
we found that counting approximately 150 to 200 points on 20 or 25
systematically sampled CT sections with 1.3-mm section thickness
and a point spacing of 0.3 cm enables volume estimation of
maxillary sinus with a CE below 5%. We thought that the proposed
2 methods can produce accurate volume estimations, providing an
objective tool for quantitative assessment of some sinus disease and
potential surgical intervention.
FIGURE 7. A BlandAltman analysis of the right MSV as measured by ellipsoid

formula versus stereological method. ELP, ellipsoidal; MSV, maxillary sinus
volume; ST, stereological.
FIGURE 8. A BlandAltman analysis of the left MSV as measured by ellipsoid

formula versus stereological method. ELP, ellipsoidal; MSV, maxillary sinus
volume; ST, stereological.
e43
Study Limitations
This study has 2 limitations. The first one is the small number of
subjects in age groups. Larger populations will be investigated for
more reliable and accurate statistical results in future studies. The
second one is that used methods which are manual. Automatic
methods need to be evaluated which will be performed in a clinical
study comparing all parameters.
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A Novel Technique for Short

Nose Correction: Hybrid Septal
Extension Graft
Jong Seol Woo, MD, Nguyen Phan Tu Dung, MD PhD,y
and Man Koon Suh, MD
Background: There are many techniques for correcting short nose
deformities and the septal extension graft is the most commonly
#
performed technique among Asians. In many Asian patients septal

cartilage, however, is too small and insufficient to perform an
effective septal extension graft. Therefore, we designed a novel
technique, named hybrid septal extension graft to overcome this
pitfall in Asian tip plasty.
Methods: From February 2010 to March 2013, 41 patients with
primary (N 30) or secondary (N 11) short nose deformity
underwent a hybrid septal extension graft. The hybrid septal
extension graft is a modified septal extension graft which uses
the small septal cartilage along with irradiated homologous costal
cartilage. Irradiated homologous costal cartilage was carved into a
shape of a thin batten and securely fixed bilaterally to the caudal
septum. Harvested septal cartilage was located between the 2 irradiated homologous costal cartilage batten grafts and fixed with
sutures. Then, the alar cartilage was fixed at the end of the septal
cartilage graft. The nasal lengths, nasal tip projections, and nasolabial angles were measured pre- and postoperatively.
Results: The hybrid septal extension graft showed enough nose
lengthening and a decreased nostril show, even in cases with a very
small septal cartilage.
Conclusions: The authors present a novel technique for correction
of short nose deformity in Asians. The hybrid septal extension graft
provides good results with minimal complications and overall
patient satisfaction was very high.
Key Words: Asians, hybrid septal extension graft, short nose
orrection of short nose deformity consists of several important

steps, such as release of alar cartilage from the upper lateral
cartilage, wide undermining and release of dorsal skin flap, and
fixation of lengthened alar cartilage. For the fixation of lengthened alar cartilage, septal extension graft is one of the most secure
and most commonly performed techniques. Harvested septal
cartilage should be more than 25 mm in length, although this
may vary according to the needs of the patient. Most Asian
patients have very small and insufficient septal cartilage which
makes it difficult to be used as an effective septal extension graft.
For this reason, autogenous rib cartilage or irradiated homologous
costal cartilage (IHCC) may be used as an alternative. These
alternatives, however, have disadvantages. An autogenous rib
cartilage may evoke the patients worries about a scar on the chest
wall and they are often hesitant to go under general anesthesia.
Moreover, an autogenous rib cartilage or IHCC may lead to a very
rigid nasal tip.1,2
The hybrid septal extension graft is a modified technique of
septal extension graft that can use even a very small septal cartilage
for septal extension by using IHCC or conchal cartilage simultaneously to supplement the small septal cartilage. In this study, we
describe a novel technique of the hybrid septal extension graft for
correcting short nose deformities in Asian patients.
From the JW Plastic Surgery Center, Seoul, South Korea; and yJW Plastic
Surgery Vietnam Clinic, Ho Chi Minh City, Vietnam.
Received June 16, 2015.
Accepted for publication August 16, 2015.
Address correspondence and reprint requests to Man Koon Suh, MD, JW
Plastic surgery Center, Samsin Building, 836 Nonhyeon-ro, Gangnamgu, Seoul 135-893, South Korea; E-mail: smankoon@hanmail.net
The authors report no conflicts of interest.
ISSN: 1049-2275
DOI: 10.1097/SCS.0000000000002307
#
MATERIALS AND METHODS

Patients
From February 2010 to March 2013, 41 patients, both women
(N 38) and men (N 3), with a different degree of short nose
underwent a hybrid septal extension graft. All 41 patients required
septal extension graft for short nose correction. Because their septal
cartilages were too small to be used as a septal extension graft, we
performed a hybrid septal extension graft using autogenous septal
cartilage combined with IHCC in 39 patients and a conchal cartilage
in 2 patients. The patients were divided into 30 primary cases and
11 secondary cases. Combined operations consisted of dorsal
augmentation with silicone implant (N 35), Gore-tex (N 2),
and dermofat (N 3). Corrective rhinoplasty was performed in
2 patients with deviated nose.
METHODS
Photometric Evaluation
We used proportional indices that were described by previous
report of Kim et al3 to evaluate the postoperative outcomes. Pre- and
postoperative lateral views were obtained from each patient and the
glabella, sellion, subnasale, pronasale, and pogonion were identified.
The proportional indices, such as nasal bridge length index and nasal
tip projection index, were measured (Fig. 1). Also the columellalabial angle was measured. All indices were obtained before and after
surgery (Table 1). We used the paired t-test to compare differences in
these values before and after surgery. The statistical analyses were
performed by SPSS (version 19.0, IBM, Armonk, NY).
Modified Septal Extension Grafting (Hybrid

Septal Extension Graft)
All patients were operated under local anesthesia with intravenous sedation using propofol and midazolam.
Open rhinoplasty technique was performed as follows; an
inverted V-shape transcolumella incision or incision along the
previous open rhinoplasty incision scar was made and was extended
upward along the anterior margin of the medial crus, caudal margin
of alar dome area and then laterally extended along the caudal
margin of the lateral crus. In primary cases, dissection was done
above the supraperichondrial plane and the alar cartilage was fully
FIGURE 1. The points and measured indices. Glabella, the most prominent
point in the midline between the brows; sellion, the deepest point of the
nasofrontal angle at the intersection of forehead slope and nasal slope;
pronasale, the most prominent point on the nasal tip; subnasale, the point
beneath the nose where the columella merges with the upper lip in the
midsagittal plane; and pogonion, the most anterior point on the chin.
e45
TABLE 1. Patient Information

Length Index
Case
1
2
3
4
5
6
7
8
9
10
11
12
13
14
15
16
17
18
19
20
21
22
23
24
25
26
27
28
29
30
31
32
33
34
35
36
37
38
39
40
41
Projection Index
Nasolabial Angle
Age (year)
Sex
Follow-up (Month)
Preop
Postop
Preop
Postop
Preop
Postop
21
37
35
23
27
41
31
33
20
29
53
34
27
19
35
26
36
42
23
37
33
58
28
35
30
24
43
38
45
22
19
23
34
32
58
30
29
27
40
25
33
F
F
F
F
F
F
F
F
F
M
F
F
F
F
M
F
F
F
F
F
F
F
F
F
F
F
F
F
F
F
F
F
F
F
F
F
F
M
F
F
F
16
12
14
36
12
24
15
12
12
14
13
12
16
12
13
23
15
12
36
12
18
13
12
13
20
16
12
24
14
12
12
12
18
14
12
34
21
12
15
12
14
33.33
31.58
30.77
31.69
32.05
31.38
33.16
31.37
31.74
32.63
30.92
31.86
31.54
31.77
32.47
33.06
32.75
31.77
30.56
31.23
31.63
31.28
31.26
30.76
32.36
31.52
31.48
32.33
31.29
30.86
33.16
32.05
31.72
30.58
31.89
31.32
31.07
30.95
31.61
31.27
32.19
36.84
38.71
37.29
35.93
37.32
36.85
38.03
37.42
38.03
37.82
36.37
36.31
36.88
37.13
36.95
38.34
37.29
36.01
36.42
36.97
37.31
38.04
36.75
59.92
37.18
36.82
38.62
37.48
38.31
37.25
37.03
38.03
38.36
37.71
38.67
36.78
38.42
37.47
36.28
37.79
38.32
7.89
8.77
8.46
8.39
7.52
7.9
8.95
8.42
8.51
8.06
8.47
8.12
8.64
8.83
8.19
8.05
7.62
8.71
8.54
8.69
8.26
8.17
7.83
8.42
8.29
8.34
8.65
8.61
8.9
8.34
8.79
8.41
8.53
7.71
8.29
8.74
8.58
8.45
8.27
8.07
7.94
10.53
11.29
11.02
10.93
11.23
10.61
11.34
10.38
11.27
10.78
11.23
11.03
11.45
11.26
10.66
10.13
10.51
11.2
11.16
11.05
10.48
10.77
11.12
11.37
10.93
11.28
10.97
11.33
11.15
11.2
11.16
10.74
11.04
10.85
11.36
11.52
11.29
10.86
11.29
11.16
11.24
113
104.5
109
105
106.5
108.5
106
103.5
108
106
110.5
107
109
107.5
111
106.5
109.5
107
107.5
108.5
109.5
105
109
105.5
107
107.5
110.5
106.5
108.5
106.5
104.5
105.5
109.5
106
107.5
111.5
106.5
107
104
105.5
106
97
94
89.5
93.9
92
96.5
92.5
94
90.5
94.5
92.5
97.5
97
99.5
96.5
98
96.5
93
91.5
94
93.5
96.5
96.5
97.5
94.5
95
96.5
94.5
96
93.5
93
96
95.5
96.5
97.5
96
93.5
96.5
91.5
93.5
95
M, male; F, Female; Preop, preoperative; Postop, postoperative.
exposed. Subperiosteal dissection over the nasal bone was done

when a dorsal augmentation was planned. Wide subperiosteal and
supraperichondrial dissection was performed to release and
lengthen the skin envelope. Transverse nasalis muscle was released
bilaterally at the pyriform aperture to further lengthen the skin
envelope. Then, the lower lateral cartilage was released for its
caudal repositioning. First, the scroll area between the upper and
lower lateral cartilage was released by a Metzenbaum scissors. This
release was done so that the thin whitish vestibular mucosa alone
was left between the upper and lower lateral cartilages. Also, a
disconnection of the accessory ligament was done if the lengthening
of the lower lateral cartilage was not sufficient with the scroll area
dissection alone. In addition, dissection of membranous septum was
sometimes necessary for a more caudal release of the lower lateral
cartilage. In secondary rhinoplasty, the release of lower lateral
cartilage was not different from that of primary case, even though
the abundant scar tissue usually makes it more difficult and time
consuming. Dual plane dissection was carried out to lengthen the
e46
skin envelope. After elevating the scar tissue and the skin envelope,
we separated the skin envelope from the underlying scar tissue or
capsule, allowing the skin to lengthen and cover the extended alar
cartilage. The septal cartilage was harvested leaving 10 to 12 mm of
L-strut, depending on the strength of the septum.
The carved IHCC, approximately 1.0 mm in thickness and 10 to
15 mm in length, was grafted on both sides of the caudal septum
(batten type) (Fig. 2A). Ear cartilage could be used as an alternative
to IHCC. 50 PDS anchoring sutures were used in 3 to 4 locations
to fixate the grafts firmly. The harvested small septal cartilage was
located between the 2 IHCC batten grafts and rigidly fixated using
50 PDS (Fig. 2B). The tip tripod was then caudally pulled for
fixation with 50 PDS sutures between the bilateral alar domes and
the anterior edges of the septal cartilage graft (Fig. 3A). If the
harvested septal cartilage was exceptionally small (less than 10 mm
length), it was pulled more caudally between the IHCC grafts,
without touching the caudal septum (Fig. 3B). Tip onlay graft or
shield graft was performed for further tip projection, if necessary.
#
FIGURE 2. Thecarvedirradiatedhomologous costalcartilage(A) andthe irradiated

homologous costal cartilage located in both sides of the caudal septum (B).
FIGURE 4. Cases of rhinoplasty with hybrid septal extension graft. Case 1a,
preoperative and 1b, 18-month postoperative (left). Case 2a, preoperative and
2b, 12-month postoperative (center). Case 3a, preoperative and 3b, 30-month
postoperative (right).
DISCUSSION
FIGURE 3. The hybrid septal extension graft. A, The harvested septal cartilage is
located between the 2 irradiated homologous costal cartilage batten grafts. B, If
the harvested septal cartilage is exceptionally small, septal cartilage can be fixed
with irradiated homologous costal cartilage without touching the caudal septum.
Dorsal augmentation was done using silicone implants or GoreTexR with 2.0 to 4.0 mm thickness. Silicone wrapped by deep
temporal fascia or dermofat graft harvested from buttock was used
in patients who presented with thin skin.
RESULTS
The range of nasal lengthening in 41 patients was between 3 to
10 mm (mean, 5.6 mm). There were no perioperative complications,
such as infection, resorption, warping or asymmetric tip or columella. All patients were followed up for an average of 15 months
(range 12 months to 3 years). No major complications associated
with the implants, such as exposure or migration of the implants,
necrosis of the overlying tissues, or infection caused by the implants
were noted during the follow-up period. Three cases needed minor
revision because of inadequate lengthening and tip projection. Two
patients complained of high tip projection whereas 1 patient
complained of low tip projection. Trimming of septal cartilage
and cephalic rotation of alar cartilage were performed to correct the
high projected tip. An onlay graft using the conchal cartilage was
performed to correct low tip projection.
There were statistically significant differences between the preand postoperative values in nose length and nose tip projection. Also,
the nasolabial angle was significantly reduced (Table 2). Most patients
were satisfied with their nasal contour and tip projection (Fig. 4).
TABLE 2. The Results of the Photometric Evaluation of Indices
Length index
Projection index
Nasolabial angle index
Preop (N 41)
Postop (N 41)
31.70 0.7
8.40 0.4
107.40 2.1
37.90 3.6
11.00 0.3
94.9 2.2
<0.001
<0.001
<0.001
Preop, preoperative; Postop, postoperative.
In this study, the hybrid septal extension graft for short nose patients
showed enough nasal lengthening and tip projection, even in
patients with a very small septal cartilage. Autogenous cartilages,
such as septal and conchal cartilages are optimal as graft materials
for nasal tip projection and derotation.4 7 In short nose corrections,
the septal cartilage is most commonly used for the septal extension
graft.8,9 Septal cartilage is used most commonly as donors because
it can directly extend and strongly support the alar cartilage and it
can be harvested easily in the same operative filed.10
Septal extension graft is an effective procedure for tip projection
and lengthening during rhinoplasty. Septal extension graft was first
reported by Byrd et al11 which was classified according to the
stability of the caudal septum and the amount of septal cartilage.
Subsequently, a few modified techniques were reported such as
tongue-and-groove technique by Guyuron et al12 and extensive
harvest technique by Kim et al.3 In these techniques, however, large
amount of septal cartilage is needed.
In Asians, however, the adequate septal cartilage harvesting is
not always possible because of insufficient quantity, deviation,
weakness, and severe ossification.13 A septal extension graft for
short nose correction needs a septal cartilage of more than 20 mm in
length. The mean septal cartilage which could be harvested was
12.1 mm 18.0 mm if the remained L-strut was of 10 mm width in a
Korean cadaver study by Kim et al.3 The harvested septal cartilage
however, is usually too small to be used as an effective septal
extension graft. Moreover, in these patients, more septal cartilage
should be preserved as an L-strut for the stability of the nasal
framework.9 In these cases, ear cartilage or rarely rib cartilage can
be used for the septal extension graft. Even though ear cartilage can
be used for septal extension, its predictability is somewhat low
because of its size and curvature. Rib cartilage on the contrary, is
abundant, durable, and stronger than septal cartilage. Rib cartilage,
however, is not widely used as it is a more invasive procedure
requiring general anesthesia and warping is one of the major
disadvantages of autogenous rib cartilage grafts.2
We applied a new technique to overcome a very small septal
cartilage as a troublesome obstacle in Asian short nose correction. It is
called hybrid septal extension graft and requires only 10 to 15 mm
length of septal cartilage. With this technique it is possible to achieve
sufficient tip projection and extension. We named this new technique
as hybrid septal extension graft (hybrid SEG), as it uses 2 different
kinds of cartilage for the modified type of septal extension graft.
Hybrid SEG uses the IHCC or conchal cartilage as a bilateral batten
graft fixed to caudal septum and it provides more extension to caudal
septum and provides basis for septal extension grafts. Harvested small
septal cartilage is fixed between the bilateral IHCC (or conchal
cartilage), even a small septal cartilage can act as a strong septal
extension graft based on the lengthened caudal septum.
e47
This hybrid SEG has several advantages: the nasal tip is softer
and mobile than in cases using autogenous rib cartilage or IHCC
alone. Compared to the conventional septal extension the graft
deviation is much less, since septal cartilage is centrally located
between the 2 IHCC or ear cartilage in hybrid SEG. The grafted tip
receives pressure from the alar cartilage in a very parallel direction
compared with the unilaterally fixed graft. Therefore, a chance of
nasal tip deviation is extremely low for these 2 reasons. Compared
to IHCC use alone, a deformity or relapse because of this technique
would be much smaller in case the IHCC would be unexpectedly
resorbed, because hybrid SEG uses a smaller amount of IHCC than
the septal extension graft using IHCC only. If the resorption rate is
the same, a deformity would be smaller in case of using less volume
of IHCC than in case of using a larger volume. Recently, there are
many reports about the IHCC graft for rhinoplasty. Irradiated
homologous costal cartilage is easy to manipulate and has low
donor site morbidity compared to autogenous rib cartilage. And
multiple grafts are possible because of its abundant quantity.
Therefore, the IHCC graft is increasingly used as graft material
especially in patients with short nose deformities with limited donor
because of previous operation. The use of IHCC grafts, however, is
still controversial in terms of resorption, warping, and infections.10
Some papers show that the absorption rate is not significantly different
from autogenous cases,1416 but other reports insist that 100% of the
IHCC graft were completely absorbed.17 Suh et al1 reported minimal
2-year follow-up cases where the use of IHCC solemnly showed no
sign of resorption and its original shape was maintained. They
recommended to select the dense area of the IHCC and to do a tight
fixation of the grafts. Warping may occur immediately or delayed
after cartilage graft. In short nose patients, delayed warping of
cartilage graft may be more common because of skin tension. To
avoid immediate and delayed warping, we waited at least for 1 hour
after cutting the cartilage before insertion to check for immediate
warping of cartilage. To minimize delayed warping, we grafted
cartilage bilaterally and folded the cartilage onto the same surface.
Commercially supplied IHCC does not have an intrinsic uniform
density; some area is dense while the other area is crumbly. The
crumbly area is a degenerative area and has no visible lacunae or
chondrocyte, even though the chondrocyte itself inside the IHCC is
not a living cell. During hybrid SEG, we selected only the dense
area of the IHCC and rigid fixation sutures were done in more than 3
points between the bilateral grafts and the septal cartilage graft. We
used a minimal size of IHCC as bilateral batten grafts that acted as a
bridge to locate the septal cartilage in a stable and proper position
for the septal extension graft. It may cause less deformity of the
nasal tip even if there is any unexpected resorption of IHCC,
because IHCC is less contributable to septal extension in hybrid
SEG than in SEG using IHCC alone. The use of ear cartilage instead
of IHCC may cause much less worries about resorption.
There are many methods to measure the outcome after rhinoplasty.1820 It is hard to convince all patients to regularly visit the
hospital and to measure indices directly even though it is the best way
to estimate the outcome of surgery. Therefore, we took clinical
pictures of the lateral views of the patients before and after rhinoplasty
and measured the change of tip projection and nasal length on the basis
of 2 points; glabella and pogonion (Fig. 1). In the photometric
evaluation, the projection of nasal tip and nasal length were significantly increased and the columellar-labial angle was also significantly
decreased in patients who underwent hybrid SEG.
CONCLUSIONS
Small septal cartilage is the most common obstacle a surgeon must
overcome during short nose corrections in Asian patients. With our
novel technique, surgeons may effectively lengthen the nose even in
patients with small septal cartilage.
e48
REFERENCES
1. Suh MK, Ahn ES, Kim HR, et al. A 2-year follow-up of irradiated
homologous costal cartilage used as a septal extension graft for the
correction of contracted nose in Asians. Ann Plast Surg 2013;71:4549
2. Kim SK, Kim HS. Secondary Asian rhinoplasty: lengthening the short
nose. Aesthet Surg J 2013;33:353362
3. Kim JS, Han KH, Choi TH, et al. Correction of the nasal tip and columella
in Koreans by a complete septal extension graft using an extensive
harvesting technique. J Plast Reconstr Aesthet Surg 2007;60:163170
4. Sheen JH, Sheen AP. Aesthetic Rhinoplasty. 2nd ed. St Louis, MO:
Quality Medical Publishing; 1998
5. Marin VP, Landecker A, Gunter JP. Harvesting rib cartilage grafts for
secondary rhinoplasty. Plast Reconstr Surg 2008;121:14421448
6. Jang YJ, Yu MS. Rhinoplasty for the Asian nose. Facial Plast Surg
2010;26:93101
7. Gunter JP, Rohrich RJ. Lengthening the aesthetically short nose. Plast
Reconstr Surg 1989;83:793800
8. Lin J, Chen X, Wang X, et al. A modified septal extension graft for the
Asian nasal tip. J Am Med Assoc Facial Plast Surg 2013;15:362368
9. Paik MH, Chu LS. Correction of short nose deformity using a septal
extension graft combined with a derotation graft. Arch Plast Surg
2014;41:1218
10. Cochran CS, Gunter JP. Secondary rhinoplasty and the use of
autogenous rib cartilage grafts. Clin Plast Surg 2010;37:371382
11. Byrd HS, Andochick S, Copit S, et al. Septal extension grafts: a
method of controlling tip projection shape. Plast Reconstr Surg
1997;100:9991010
12. Guyuron B, Varghai A. Lengthening the nose with a tongue-and-groove
technique. Plast Reconstr Surg 2003;111:15331539
13. Jeong JY. Obtaining maximal stability with a septal extension technique
in East Asian rhinoplasty. Arch Plast Surg 2014;41:1928
14. Lefkovits G. Irradiated homologous costal cartilage for augmentation
rhinoplasty. Ann Plast Surg 1990;25:317327
15. Demirkan F, Arslan E, Unal S, et al. Irradiated homologous costal
cartilage: versatile grafting material for rhinoplasty. Aesthetic Plast Surg
2003;27:213220
16. Kridel RW, Ashoori F, Liu ES, et al. Long-term use and follow-up of
irradiated homologous costal cartilage grafts in the nose. Arch Facial
Plast Surg 2009;11:378394
17. Welling DB, Maves MD, Schuller DE, et al. Irradiated homologous
cartilage grafts. Long-term results. Arch Otolaryngol Head Neck Surg
1988;114:291295
18. Dhong ES, Kim YJ, Suh MK. L-shaped columellar strut in East Asian
nasal tip plasty. Arch Plast Surg 2013;40:616620
19. Park JH, Mangoba DC, Mun SJ, et al. Lengthening the short nose in
Asians: key maneuvers and surgical results. JAMA Facial Plast Surg
2013;15:439447
20. Huang J, Liu Y. A modified technique of septal extension using a septal
cartilage graft for short-nose rhinoplasty in Asians. Aesthetic Plast Surg
2012;36:10281038
Rhabdomyolysis Presenting as
Orbital Apex Syndrome
Jae Min Wi, MD and Mijung Chi, MD, PhD
Abstract: Rhabdomyolysis is a condition in which striated muscle
tissue breaks down rapidly and releases muscular cell constituents
into extracellular fluid and the circulation. Renal symptoms, such as
acute renal failure, are major complications of rhabdomyolysis.
However, no previous report of rhabdomyolysis associated with
orbital complication has been issued. Here, we report the first
patient of rhabdomyolysis presenting as orbital apex syndrome.
A 66-year-old man presented with right periorbital swelling with
erythematous patches and conjunctival chemosis. In addition,
#
swelling, redness, and vesicles were observed in both lower legs. He

was found in a drunken state with the right side of his face pressed
against a table. Ophthalmic examination showed right eye fixation
in all directions and ischemic change of retina. Blood testing
showed elevated muscle enzyme associated with muscle destruction.
And computed tomography of the orbit showed swelling of right
extraocular muscles and crowding of right orbital apex. Under a
diagnosis of rhabdomyolysis-associated orbital apex syndrome and
central retinal artery occlusion, intravenous steroid and antibiotics
therapy with intraocular pressure-lowering topicals were begun.
Clinical presentation, treatment course, and follow-up are discussed.
Key Words: Central retinal artery occlusion, orbital apex
syndrome, rhabdomyolysis
FIGURE 1. External appearance of the patient. He presented with right

periorbital swelling and redness. Extraocular movement testing revealed the
right eye to be fixed in all directions.
habdomyolysis is a potentially life-threatening syndrome

characterized by the breakdown of skeletal muscle resulting
in the subsequent release of intracellular contents into the circulatory system. Renal symptoms, such as acute renal failure, are major
complications of rhabdomyolysis.
Orbital apex syndrome is a collection of cranial nerve deficits
associated with a space occupying lesion near the apex of an orbit.1
Visual loss and ophthalmoplegia are often the initial manifestations
of orbital apex syndrome, and optic atrophy typically develops over
weeks to months.1 Clinical presentation, treatment course, and
follow-up are discussed.
CLINICAL REPORT
A 66-year-old man was found in a drunken state with the right side of
his face pressed against a table. On arrival at the emergency room, his
right visual acuity was no light perception and his intraocular pressure
was 26 mm Hg. Severe right periorbital swelling with erythematous
patches and conjunctival chemosis were noted. His right pupil was
dilated to 7.0 mm and fixed. In addition, swelling, redness, and
vesicles were observed in both lower legs. Extraocular muscle movement testing showed the right eye was fixed in all directions (Fig. 1),
and fundus examination revealed disc hemorrhage and whitening of
the retina in the posterior pole, and a cherry red spot in the center of the
macula (Fig. 2A). In fluorescein angiography showed delayed choroidal filling and a severely delayed retinal arteriovenous passage
time (Fig. 2B). Blood test results were as follows: creatine
kinase >15,000 U/L (normal range 32294 U/L), lactate dehydrogenase was 1711 U/L (normal range 200485 U/L), and creatine
phosphokinase 99.01 ng/mL (normal range 05 ng/mL). These results
were indicative of muscle destruction. A computed tomography scan
of the orbit conducted at the time showed swelling of right extraocular
muscles and crowding of orbital apex (Fig. 2C, D).
Under a diagnosis of orbital apex syndrome and central retinal
artery occlusion associated with rhabdomyolysis, high-dose intravenous methylprednisolone injection (500 mg q12hrs for 3 days)
with intraocular pressure lowering topicals was administered. He
From the Department of Ophthalmology, Gachon University Medicine and
Science Gil Hospital, Incheon, Republic of Korea
Received July 15, 2015.
Accepted for publication October 1, 2015.
Address correspondence and reprint requests to Mijung Chi, MD, PhD,
Department of Ophthalmology, Gachon University Medicine and
Science Gil Hospital, 1198, Guwol-dong, Namdong-ku, Incheon 405760, Republic of Korea; E-mail: cmj@gilhospital.com
ISSN: 1049-2275
DOI: 10.1097/SCS.0000000000002308
#
FIGURE 2. Fundus revealing the aspect of central retinal artery occlusion

(A), fluorescein angiography revealing remarkable slowing of dye flow (B),
orbital computed tomography showing swelling of right extraocular muscles
(C) and crowding of orbital apex (D), atrophic, pale disc at final follow-up (E).
was also treated medically with third-generation intravenous antibiotics and fluid administration. During the third month of followup, the swelling and redness and vesicles in the right periorbital area
(Fig. 3) and in both lower legs were improved, and blood testing
FIGURE 3. During the third month of follow-up, swelling and redness and
vesicles in the right periorbital area improved, but extraocular muscle
movement testing revealed continued right eye fixation in all directions.
e49
FIGURE 4. During the sixth month of follow-up, ischemic skin changes on the
right periorbital area had fully recovered and right eye movement had
significantly improved.
showed the levels of enzymes associated with muscle destruction

had decreased to normal values. However, his right visual acuity
was still no light perception, and the right eye fixation persisted.
During the sixth month of follow-up, right eye movement was
significantly improved (Fig. 4), but right visual acuity remained at
no light perception. Furthermore, a fundus examination revealed the
right disc had become atrophic and pale (Fig. 2E). The patient is
now under outpatient follow-up without additional treatment.
pressure-lowering topicals was applied to address orbital apex syndrome. During the third month of follow-up, ischemic skin change on
the right periorbital area and both lower legs were improved, and
enzymes in blood associated with muscle destruction had decreased to
normal values. Nevertheless, right visual acuity remained at no light
perception and right eye fixation persisted. During the sixth month of
follow-up, right eye movement was improved, but right visual acuity
showed no improvement, and the disc had become atrophic and pale.
Generally, rhabdomyolysis is commonly complicated by renal
disease, which is attributed to renal tubular injury caused by
myoglobin release from muscle cells.3,4 Hence, the primary therapeutic goal of rhabdomyolysis is usually to prevent renal complications, and intravenous antibiotics and fluid administration are
usually administered for this purpose.
The prognosis of alcohol-induced rhabdomyolysis is relatively
benign.2,3 In our patient, ischemic change on the right periorbital area
and both lower legs improved, and blood testing showed enzymes
associated with muscle destruction and renal function decreased to
normal values. On the other hand, visual acuity was not improved.
To the best of our knowledge, this is the first patient of rhabdomyolysis manifesting as orbital apex syndrome. This case cautions
ocular and renal complications should be considered when dealing
with rhabdomyolysis, and that when orbital apex syndrome is
encountered, rhabdomyolysis should be considered a possible cause.
REFERENCES
DISCUSSION
Rhabdomyolysis involves the disintegration of striated muscle, which
causes the release of muscular cell constituents into the extracellular
fluid and the circulation, and subsequent complications.2
The causes of rhabdomyolysis are classified as traumatic or
nontraumatic.2 Nontraumatic causes include muscular disorders,
electrolyte abnormalities, fever, infections, heavy exercise, epilepsy,
and drug taking.2 The common drugs that cause rhabdomyolysis are
alcohol, cocaine, amphetamine, and lovastatin.2,3 In the described
patient, alcohol intoxication is regarded as main etiology. Alcohol can
cause rhabdomyolysis via a direct myotoxic effect or inducing
metabolic abnormalities, such as hypophosphatemia and hypokalemia.46 Alcohol can also predispose the individual to trauma, seizures,
or coma-induced ischemic pressure necrosis.24 Rhabdomyolysis
associated with alcohol intoxication can manifest in degrees ranging
from a subclinical rise in creatine kinase to a medical emergency due
to interstitial and muscle cell edema, contraction of intravascular
volume, and pigment-induced acute renal failure.3,4 Today, rhabdomyolysis is a leading cause of acute renal failure.24 However, in our
patient, it manifested as orbital apex syndrome, which results from a
collection of cranial nerve deficits associated with a space-occupying
lesion near the apex of an orbit.1 Visual loss and ophthalmoplegia are
often the initial manifestations of orbital apex syndrome, and optic
atrophy typically develops over weeks to months.1 The causes of
orbital apex syndrome are classified as inflammatory, infectious,
neoplastic, and vascular.1 However, rhabdomyolysis has not been
previously reported to be a cause of orbital apex syndrome. In our
case, the patient was found in drunken state while prone, and
prolonged compression of the right orbital area and both lower legs
may have induced ischemic changes and resulted in muscle destruction. Furthermore, muscle cell death initiates a vicious cycle of
increased capillary permeability and muscle swelling.7 In our patient,
orbital computed tomography revealed swelling of right extraocular
muscles, and narrowing of the right orbital apex. Under the influence
of this vicious cycle, increased pressure on the orbital apex is believed
to cause orbital apex syndrome manifesting as visual loss with
central retinal artery occlusion and ophthalmoplegia. In our
patient, high-dose intravenous methylprednisolone with intraocular
e50
1. Yeh S, Foroozan R. Orbital apex syndrome. Curr Opin Ophthalmol

2004;15:490498
2. Vanholder R, Sever MS, Erek E, et al. Rhabdomyolysis. J Am Soc Nephrol
2000;11:15531561
3. Shin WS, Kim YO, Han CM, et al. The clinical features of acute renal
failure caused by alcohol induced rhabdomyolysis. Korean J Nephrol
1999;18:17
4. Poels PJ, Gabreels FJ. Rhabdomyolysis: a review of the literature. Clin
Neurol Neurosurg 1993;95:175192
5. Rubin E, Katz AM, Lieber CS, et al. Muscle damage produced by chronic
alcohol consumption. Am J Pathol 1976;83:499516
6. Song SK, Rubin E. Ethanol produces muscle damage in human
volunteers. Science 1972;175:327328
7. Dolberg-Stolik OC, Putterman C, Rubinow A, et al. Idiopathic capillary
leak syndrome complicated by massive rhabdomyolysis. Chest
1993;104:123126
Transverse Slicing of the

SixthSeventh Costal
Cartilaginous Junction:
A Novel Technique to Prevent
Warping in Nasal Surgery
Tara Lynn Teshima, MSc, FRCSC, Homan Cheng, MD,y
Amir Pakdel, PhD,y Alex Kiss, PhD,z and
Jeffrey A. Fialkov, MD, FRCSCy
Background: Costal cartilage is an important reconstructive tissue
for correcting nasal deformities. Warping of costal cartilage, a
recognized complication, can lead to significant functional and
aesthetic problems. The authors present a technique to prevent
warping that involves transverse slicing of the sixthseventh costal
#
cartilaginous junction, that when sliced perpendicular to the long

axis of the rib, provides multiple long, narrow, clinically useful
grafts with balanced cross-sections. The aim was to measure
differences in cartilage warp between this technique (TJS) and
traditional carving techniques.
Methods: Costal cartilage was obtained from human subjects and
cut to clinically relevant dimensions using a custom cutting jig. The
sixthseventh costal cartilaginous junction was sliced transversely
leaving the outer surface intact. The adjacent sixth rib cartilage was
carved concentrically and eccentrically. The samples were incubated and standardized serial photography was performed over time
up to 4 weeks. Warp was quantified by measuring nonlinearity of
the grafts using least-squares regression and compared between
carving techniques.
Results: TJS grafts (n 10) resulted in significantly less warp than
both eccentrically (n 3) and concentrically carved grafts (n 3)
(P < 0.0001). Warp was significantly higher with eccentric carving
compared with concentric carving (P < 0.0001). Warp increased
significantly with time for both eccentric (P 0002) and concentric
(P 0.0007) techniques while TJS warp did not (P 0.56).
Conclusion: The technique of transverse slicing costal cartilage
from the sixthseventh junction minimizes warp compared with
traditional carving methods providing ample grafts of adequate
length and versatility for reconstructive requirements.
technically difficult and still carry with them a significant amount of

unpredictability for persistent warping and other complications
such as resorption in the case of irradiated cartilage and hardware-related complications in the case of K-wire fixation. To date,
concentrically carved cartilage grafts have been the gold standard
for the prevention of warp in nasal reconstruction.
The sixthseventh costal cartilaginous junction, or synchondrosis, is a unique anatomic structure in the thorax that extends from
the sixth to the seventh cartilaginous rib just lateral to the costal
margin. While concentric carving of costal cartilage requires grafts
to be carved parallel to the long axis of the rib, in order to yields
grafts of adequate length for clinical use, the sixthseventh costochondral junction is of dimensions that when sliced transversely and
perpendicular to the long axis of the junction itself yield long
narrow grafts of any desired thickness. In this way, these grafts can
be harvested in their final dimensions without having to breach the
outer surface of the costo-chondral junction, thus maintaining a
cross-sectional balance of forces. Multiple transverse slices are
obtained giving an abundance of cartilage grafts of various lengths
and controlled thickness ideal for nasal reconstruction. We have
used this novel slicing technique for reconstructive and secondary
rhinoplasty, producing grafts of ideal dimension used for septal
extension, spreader, columellar strut, rim, and batten grafts with
reliable long-term results. On the basis of our clinical experience
with this new technique, we hypothesized that transverse slicing of
the sixthseventh costal cartilaginous junction will lead to a
significantly decreased incidence of warping when compared with
conventional concentric carving.
Key Words: Cartilage grafts, cartilage warp, costal cartilage, nasal

reconstruction, prevent warp, sixthseventh costal cartilaginous
junction, transverse slicing
ostal cartilage is an important reconstructive tool for correcting

nasal deformity resulting from cancer resection, trauma, and
congenital deformities. It is also an invaluable source of cartilage in
secondary aesthetic rhinoplasty. The major drawback of the use of
costal cartilage graft is its tendency to warp. Warping of costal
cartilage can lead to functional airflow problems and cosmetic
deformity requiring subsequent surgical correction and added cost.
Warping and its consequent deformity can therefore significantly
affect the quality of life of patients requiring nasal reconstruction.
Unfortunately, there has been little advancement in the understanding and prevention of costal cartilage warping to date.
The classic studies on cartilage graft warp by Gibson and Davis1
and Fry2 postulated that warp could be minimized by balancing
cross sections. Since the discovery of what is now referred to as the
interlocking stresses, a number of techniques have been proposed to
minimize warping in an effort to make outcomes of costal cartilage
grafting more reliable.1,2 These include concentric carving, k-wire
stabilization, irradiation, and lamination.3 6 These methods can be
From the Division of Plastic Surgery, Markham Stouffville Hospital,
Markham; yDivision of Plastic Surgery, Sunnybrook Health Sciences
Centre, University of Toronto; and zDepartment of Biostatistics, Sunnybrook Health Sciences Centre, Toronto, Canada.
Address correspondence and reprint requests to Dr Jeffrey A. Fialkov, MD,
FRCSC, Division of Plastic and Reconstructive Surgery, Department of
Surgery, University of Toronto, M1 516 Sunnybrook Health Sciences
Centre, Toronto, ON M4N 3M5, Canada;
E-mail: jeff.fialkov@sunnybrook.ca
ISSN: 1049-2275
DOI: 10.1097/SCS.0000000000002309
#
METHODS
Patients
Costal cartilage from the sixthseventh costal cartilaginous
junction was obtained from patients treated at Sunnybrook Health
Sciences Centre at the University of Toronto. Our Research Ethics
Board approved the study protocol and informed consent was
obtained from each patient. Cartilage was only harvested when
surgically indicated. It was prepared and utilized in the usual
fashion for nasal surgery, the perichondrium was not included.
Cutting Device
To obtain consistent pieces of cartilage with uniform thickness
we designed a double-blade cutting device that uses stiff, industrial
strength, single bevel blades (American Cutting Edge, Centerville,
OH).7 The blades are placed parallel to each other into slots
sectioned incrementally 1 mm apart with bevels pointed outward.
The length of each blade and therefore the maximum cartilage
length obtained was 5 cm. Cartilage thickness of 2 mm was obtained
which we felt was thickness clinically most relevant for reconstruction. The blades were secured into the cutting device using a
fastener that ensured equal uniform pressure along the length of
the blade and prevented blade flexure and movement. Stabilization
of the cartilage was done by either hand instrumentation or tension
sutures depending on what segment of cartilage was being cut. The
cutting jig was designed so the force applied to the blades was
equally distributed during the cutting process (Fig. 1).
Surgical Technique of Harvesting of the Sixth

Seventh Costal Cartilaginous Junction and
Transverse Cutting
With the patient under general anesthetic, the sixthseventh
costal cartilage junction was palpated and marked. The surgical
incision used for access was placed at the inframammary crease to
e51
FIGURE 1. Cartilage cutting device custom designed for transverse slicing of

sixthseventh junction.
minimize visible scar (Fig. 2A). En bloc removal of the sixth

seventh costal cartilage junction combined with the sixth rib was
carried out on each patient (Fig. 2B, C). For each en block piece of
cartilage the sixthseventh junctional component was marked out
to determine the boundaries of transverse slicing (Fig. 3A). Transverse slices are marked as straight lines on the junction limited to rib
with an intact outer cortex. Theoretical tensile stresses are marked
as double-headed arrows. The junction was cut into 2-mm-thick
sections producing uniform grafts (Fig. 3C). The transverse pieces
of cartilage that fulfilled the dimensional requirements were utilized
in the patients surgical procedure and were thus excluded from the
study. The remaining samples were used in the study. The remaining
cartilage from the sixth rib was used for concentric and
eccentric carving.
Surgical Technique for Concentric and

Eccentric Carving
To compare the TJS technique to traditional carving methods,
the costal cartilage from the sixth rib was cut using concentric and
eccentric techniques. As previously described, only 1 true concentric piece can be obtained from each individual costal cartilage
segment.5 Although a few techniques have been developed to obtain
a perfect concentrically carved piece of cartilage, to ensure consistent graft thickness between groups the cutting jig was used on
the basis of marking the center of each costal cartilage rib segment.
For each end of the segment, a caliper was used to measure the total
width and height. The caliper was then used as a compass to halve
these dimensions and mark them as bisecting arcs at each end of the
rib (Fig. 3D). Where these 2 arcs intersected represented the center
core of the sixth costal cartilage. The longitudinal axis from the
center point of 1 end of the segment to the center point of the
opposite end of the segment was superimposed onto the outside of
each of the lateral surfaces of the sixth costal cartilage rib using a
marking pen and a ruler. This longitudinal axis was then used to
evenly position the blades of the cutting jig set 2 mm apart and to cut
away surrounding cartilage leaving a section centered on the central
FIGURE 3. Transverse and concentric carving techniques. (A) En bloc sixth

seventh costal cartilaginous junction. Direction of forces marked in purple ink.
(B) Cutting jig positioned for cutting a transverse piece resulting in a uniform
straight graft shown in image C. (D) Utilization of calipers to mark the central
core of the sixth cartilaginous rib. (E) Cutting jig position along the central axis
of the sixth cartilaginous rib. (F) Harvesting of concentric cartilage graft from the
sixth rib. Note how initially (time 0 minutes) this graft is straight. This same graft
later goes on to warp over time (see Fig. 5).
axis of the segment (Fig. 3E and F). The length was then cut to
match the length of the longest piece of transverse cartilage
included in the study.
The eccentric piece of cartilage was obtained using the same
technique but off of the central axis of the segment. The eccentric
piece of cartilage used for the study was taken from the side
opposite the junction. After each concentric and eccentric cartilage
graft was obtained, they were cut to the same length as the longest
transverse piece included in the study for that particular patient.
Serial Photography
Cartilage warp was measured from standardized serial photographs taken at 5, 30, and 60 minutes from the time of harvest and
weekly in the tissue culture lab for up to 4 weeks. Photographs were
taken with the samples placed cut surface down on a sterile
stainless-steel platform. The platform included a reference ruler
visible in the photographic field. To obtain standardized anterior
posterior images our camera was positioned on a tripod 12 inches
from the center of the platform at a constant height and angle so that
the principle axis was centered on the sample and parallel to the
cut surface.
Tissue Culture
Following intraoperative harvesting, cartilage samples were
placed in sterile petri dishes containing Chondrocyte Growth media
(PromoCell, Heidelberg, Germany) supplemented with 50 U of
penicillin (Gibco; Thermo Fisher Scientific, Waltham, MA)/mL and
50 mg of streptomycin (Gibco)/mL to maintain viability to simulate
in vivo implantation (Fig. 4). Samples were kept at 378C for the
duration of the study. Under sterile conditions in a fume hood,
cartilage samples would undergo a media change 3 times per week
to prevent infection and maintain growth factors. For time points at
which photographs were taken, the cartilage samples were rinsed
with PBS and placed on the sterile stainless-steel platform.
Measuring Cartilage Warp

FIGURE 2. Harvesting cartilage from the sixthseventh costal cartilaginous
junction. (A) A patient marked at the inframmary crease. (B) Photograph
showing in vivo anatomy of the sixthseventh costal cartilaginous junction.
(C) En bloc removal of the sixthseventh costal cartilaginous junction including
the sixth cartilaginous rib.
e52
To measure warp we quantified the degree of deviation of each

sample from a straight line as a single metric, denoted as its
nonlinearity. In this manner any warp relative to the transverse
plane, regardless of direction, could be quantified. An interactive
#
RESULTS
Patient Demographics
FIGURE 4. Cartilage grafts incubated in tissue culture to maintain viability for

up to 4 weeks.
software module was developed (AP) for the special purpose of

calculating this quantity for each photographed sample. All
samples were numbered and the 3 independent observers were
blinded to the carving method and analyzed all of the images
independently.
The images of cartilage samples, with the ruler in view, were first
loaded into the program. Using the graphical user interface, the
observer then performed a calibration by interactively marking a
straight line between 2 points on the ruler in the image and inputting
the length of the line on the ruler in millimeters. Subsequently, the
observer marked a series of points longitudinally at the top edge of
the cartilage, which served to define the edge boundaries of the
sample in the image. A second set of points was similarly produced
on the lower edge. The software then algorithmically produced 2
independent and nonparametric spline fits for the scatter points at
each edge. The primary purpose of producing a spline-fit was to
normalize the number and density of manually defined sampling
points across all the images. Finally, the algorithm fitted a straight
line to each spline in a total-least-squares manner, also known as the
orthogonal form of linear least-squares regression fitting. The
nonlinearity metric was defined as the root-mean-squared of
the shortest distance from each of the points in the spline-fit curves
to the fitted straight line. It follows that the minimum nonlinearity
would be expected for a perfectly straight cartilage sample (ie, the
distance between the spline and the straight line would be zero). The
standard deviation of the mean was also automatically calculated by
the software.
Descriptive statistics were calculated for all variables of interest. Continuous measures were summarized using means and
standard deviations, whereas categorical measures were summarized using counts and percentages. A repeated measures regression
analysis was run to assess changes in nonlinearity between groups
and over time. The analyses adjusted for the correlation among
observations taken on the same patient to account for interpatient
variability. That is, repeated observations from the same subject
were identified as a cluster to correctly adjust the standard errors of
the test statistics and hence adjust their resulting P values. All
analyses were carried out using SAS Version 9.3 (SAS Institute,
Cary, NC).
Three patients were included in our study and a total of 22

cartilage grafts were harvested from the sixthseventh cartilaginous
junction (Table 1). The average number of transverse pieces
obtained from each sixthseventh junction was 7.33 per patient.
Excluding the transverse pieces of cartilage that fulfilled the
dimensional requirements for the patients surgery, a total of 10
grafts remained for inclusion into our study. The length of each
cartilage piece was measured and recorded. As expected by virtue
of the carving technique, 1 concentric and 1 eccentric graft were
obtained for each patient.
TJS Has Less Warp Compared With Concentric

Carving
From the 3 patients included in this study, a total of 10 transverse
cartilage grafts from the sixthseventh junction were obtained for
analysis. The average length of the transverse grafts was 3.12 cm
(range 2.34.6 cm). Transversely sliced junctional cartilage (TJS)
showed significantly less warp (mean 0.12 0.11) for all lengths and
all time points when compared with concentrically carved (mean
0.51 0.25) and eccentrically carved (mean 0.72 0.37) grafts
(P < 0.0001). As expected, warp was also significantly higher with
eccentric carving compared with concentric carving (P < 0.0001).
To control for length, the concentric and eccentric grafts from each
patient were cut to the length of the longest attainable transverse piece
from that given patient. Following the above analysis, we then
compared warp between carving techniques using only grafts of equal
length. The lengths of the 3 longest transverse and corresponding
concentric and eccentric cartilage pieces for each patient were 3.3, 3.9,
and 4.6 cm (mean 3.93 cm) respectively. Results of a repeated
measures regression analysis (adjusting for the correlation among
observations taken on the same patient and by the same rater) collapsing over time showed significantly less warp for the transversely
sliced grafts (mean 0.19 0.17) when compared with concentrically
(mean 0.51 0.25) and eccentrically (mean 0.72 0.37) carved grafts
(P < 0.0001). Furthermore, both transverse and concentric grafts of
equal length warped significantly less than eccentric grafts of equal
length (P < 0.0001 and P 0.00003 respectively).
TJS Has Less Warp Over Time Compared

With Concentric Carving
Using the previous repeated regression analysis but now incorporating the time component, the degree of warp for each carving
technique was compared intraoperatively at 5, 30, and 60 minutes
from the time of harvest, followed by weekly measurements up to 4
weeks postoperatively. Transversely sliced cartilage from the
sixthseventh costal cartilaginous junction showed significantly
less warp when compared with both concentrically and eccentrically carved grafts of equal length at all time points. A representative photograph from 1 patient comparing the carving techniques
TABLE 1. Patient Demographics Undergoing Cartilage Harvest From the SixthSeventh Costal Cartilaginous Junction
Patient
1
2
3
Age
Sex
Surgery
18
25
25
F
F
M
Cleft lip septorhinoplasty

Total Number of
Transverse Grafts
Transverse Grafts
Included in Study
Concentric Grafts
Included in Study
Eccentric Grafts
Included in Study
6
8
8
Total 22
Average 7.33
3
3
4
Total 10
1
1
1
Total 3
1
1
1
Total 3
e53
FIGURE 6. Postoperative photos taken at 4 weeks for the 3 patients included in

the study. Note for all 3 patients included in the study the transverse cartilage
grafts remain straight to 4 weeks, whereas both eccentric and concentric grafts
show warp over time.
Cartilage Length Is Related to the Degree

of Warp
To determine if cartilage length had an effect on warp we
performed a repeated measures regression analysis (adjusting for
the correlation among observations taken on the same patient and by
the same rater as before) on all the transverse pieces included in our
study. These analyses collapse across time to see the relationship
between length and warp, and whether greater length was associated
with more warp overall. Our analysis shows there is a significant
relationship (P < 0.0001) such that for each 1-unit increase in
length, warp increased by 0.066 units on average.
DISCUSSION
FIGURE 5. (A) Intraoperative photograph of the straight concentric cartilage
graft taken immediately after harvest (time 0 min). (B) Serial photographs taken
from the same patient in (A) showing increased warp over time for both
concentric carving techniques. The transverse graft of equal length remains
straight up to 4 weeks.
over time visually demonstrates the visible warp over time of

concentric carving (the current gold standard) compared with the
novel technique of transverse junctional carving (Fig. 5). Note that
in Figure 5A the concentric piece of cartilage identified by purple
dots along the 1 edge is straight at the time harvest but after
60 minutes (Fig. 5B) it has begun to warp and continues to warp
throughout the 4 weeks. Perhaps of greatest clinical significance,
transversely sliced sixthseventh junction grafts showed significantly less warp than concentrically and eccentrically carved grafts
at 4 weeks (P 0.0012). This is exemplified by the visibly straight
appearance of the transversely sliced graft at week 4 in Figure 6.
The repeated measures regression analysis reveals a significant
correlation between the degree of warp and time for eccentric
(P 0.0002) and concentric (P 0.0007) carving techniques.
No significant correlation was found for transverse grafts. In other
words, cartilage cut using concentric and eccentric carving techniques showed significantly more warp at 4 weeks compared with
when they were initially harvested. The degree of warp for transverse pieces was not significantly different at 4 weeks than it was at
the time of initial harvest (P 0.56).
e54
For a nose to appear and function normally, it must have sufficient

underlying structural support. In the absence of nasal septal cartilage, the gold standard for grafting in the nose is autologous costal
cartilage.8 Autologous costal cartilage is flexible, resilient and
provides adequate strength. However, a major drawback of costal
cartilage is its tendency to warp.
Cartilage warp is affected by cross-sectional forces that vary
depending on the location within the rib segment. Thus, the carving
technique is a critical determinant of the likelihood of graft warp.
Segments of costal cartilage sliced from the periphery (eccentric)
warp twice as much compared with cartilage taken from the central
portions of the rib (concentrically carved).5,9,10 Unfortunately,
obtaining a perfectly concentrically carved piece of cartilage is
not easy to achieve as identical amounts of cartilage equidistant
from the cross-sectional center must be removed. In addition, there
is a significant amount of cartilage wasted from the periphery to
obtain a single graft, making rib cartilage a seemingly less abundant
supply of graft material relative to the volume harvested. Despite
these limitations, however, concentric carving is still considered the
gold standard of harvesting techniques in nasal reconstruction.
There are 2 published methods documented for obtaining concentrically carved costo-chondral grafts. One method serially
removes equal amounts from all sides until a graft of desired size
has been obtained. The second technique determines the center axis
of the rib and removes all surrounding cartilage to the desired
dimensions of the beam-like graft.5,10 The latter technique was used
in the current experiment.
Despite standardizing the concentric carving technique in this
study, the procedure proved to be technically challenging, and as
#
CONCLUSION
Transverse slicing of cartilage from the sixthseventh costal cartilaginous junction is a reliable technique for obtaining strong
flexible straight grafts with clinically ideal versatile dimensions.
This technique results in less graft warp than conventional concentric techniques yielding straight grafts rendering nasal reconstructions more reliable and predictable.
FIGURE 7. Clinical application of a graft taken from the sixthseventh costal
cartilaginous junction used as a septal extension graft.
evidenced by the variable degree of warp in the concentric group

of grafts, unpredictable in the outcome. In contrast, the technique
of transverse slicing of the sixthseventh costo-chondral junction
proved technically simple, yielded multiple grafts of clinically
desirable dimensions per harvested segment, and most importantly showed significantly less warp than concentrically carved
grafts. In fact, grossly, warp was undetectable in the transversely
sliced specimens up to and including 4 weeks post harvest
(Fig. 6).
Although the likelihood of warp in concentrically carved grafts
may be reduced by choosing a rib segment that is a straight as
possible, the technical challenge of perfect concentric carving still
imparts a degree of unpredictability to the technique that appears to
be eliminated by the transverse sixthseventh junctional technique.5,10,11 Previously published techniques that attempt to balance
cross-sectional forces by slicing standard cylindrical ribs obliquely
run the risk of imbalanced forces warping grafts unpredictably
because the cross section is not perpendicular to the long access of
the rib.12,13 Furthermore, the anatomy of the sixthseventh junction
has less width than the diameter of a cylindrical rib and therefore
will yield narrower grafts better suited to purposes such as spreader,
septal extension, and strut grafts (Fig. 7). Oblique slices of cylindrical ribs will by definition maintain the diameter of the rib as their
width and cannot be trimmed narrower as this will disrupt any
potential cross-section balance of forces. The transverse sixth
seventh junctional slicing technique is one that can be utilized with
ease in the operating room with a simple cutting blade (eg, a free
Goulian knife blade) passed through the junction perpendicular to
its central axis.
New to the literature in the current study are the relationship
between graft length and warp and the maintenance of graft cellular
viability throughout the study. Our data support a positive correlation between graft length and warp. Despite this, the degree of
warp in the transverse grafts remained clinically insignificant.
Furthermore, we confirmed cellular viability of the grafts at 4 weeks
histologically better simulating the fate of implanted cartilage
grafts. In contrast, previously published studies stored cartilage
samples in saline.14
To date, the senior author has performed over 30 complex
rhinoplasty and nasal reconstructions using this technique of transverse slicing of cartilage from the sixthseventh costal cartilage
junction. We have observed clinically that our transverse cartilage
grafts are consistently straight and have shown no signs of deviation
in long-term follow-up of our patients (unpublished data). The
current study supports these clinical observations by showing
significantly less warp in transversely carved junctional grafts
compared with concentric carving for up to 4 weeks. More importantly, there was no significant change in warp (P 0.56) for TJS
grafts over the 4-week period implying clinically reliable. The
advantages to our technique are: consistent predictable results,
multiple straight grafts with a desirable variability of dimensions,
no increased morbidity to the patient, and carving is quick and easy
(a cutting jig is not required).
#
REFERENCES
1. Gibson T, Davis WB. The distortion of autogenous cartilage grafts: its
cause and prevention. Br J Plast Surg 1958;10:257274
2. Fry H. Nasal skeletal trauma and the interlocked stresses of the nasal
septal cartilage. Br J Plast Surg 1967;20:146158
3. Dingman R, Grabb W. Costal cartilage homografts preserved by
irradiation. Plast Reconstr Surg Transplant Bull 1961;28:562567
4. Gunter JP, Cochran CS. Management of intraoperative fractures of the
nasal septal L-strut: percutaneous kirschner wire fixation. Plast
5. Kim DW, Shah AR, Toriumi DM. Concentric and eccentric carved
costal cartilage: a comparison of warping. Arch Facial Plast Surg
2006;8:4246
6. Swanepoel PF, Fysh R. Laminated dorsal beam graft to eliminate
postoperative twisting complications. Arch Facial Plast Surg
2007;9:285289
7. Foulad A, Ghasri P, Garg R, et al. Stabilization of costal cartilage graft
warping using infrared laser irradiation in a porcine model. Arch Facial
Plast Surg 2010;12:405411
8. Mingrone MD, Lovice DB, Toriumi DM. Alloplastic or homograft
implantation for nasal reconstruction. In: Pensak ML, ed. Controversies
in Otolaryngology. Vol. 31. New York, NY: Thieme; 2001
9. Harris S, Pan Y, Peterson R, et al. Cartilage warping: an experimental
model. Plast Reconstr Surg 1993;92:912915
10. Adams WP Jr, Rohrich RJ, Gunter JP, et al. The rate of warping in
irradiated and nonirradiated homograft rib cartilage: a controlled
comparison and clinical implications. Plast Reconstr Surg
1999;103:265270
11. Farkas JP, Lee MR, Lakianhi C, et al. Effects of carving plane, level of
harvest, and oppositional suturing techniques on costal cartilage
warping. Plast Reconstr Surg 2013;132:319325
T, Aydin E, et al. The oblique split method: a novel
12. Tastan E, Yucel O
technique for carving costal cartilage grafts. JAMA Facial Plast Surg
2013;15:198203
13. Tastan E, Sozen T. Oblique split technique in septal reconstruction.
Facial Plast Surg 2013;29:487491
14. Foulad A, Ghasri P, Garg R, et al. Stabilization of costal cartilage graft
warping using infrared laser irradiation in a procine model. Arch Facial
Plast Surg 2010;12:405411
Trigeminal Neuralgia Caused by

Cerebellopontine Angle
Arteriovenous Malformation
Treated With Gamma
Knife Radiosurgery
Semra Isik, MD, Murat Sakir Eksi, MD,y Baran Yilmaz, MD,z
Zafer Orkun Toktas, MD,z Akin Akakin, MD,z and
Turker Kilic, MD, PhDz
Abstract: Trigeminal neuralgia is a facial pain syndrome
characterized as sudden onset and lightening-like sensation over
somatosensorial branch(es) of fifth cranial nerve. Rarely, some
e55
underlying diseases or disorders could be diagnosed, such as

multiple sclerosis, brain tumors, and vascular malformations.
The authors present a 47-year-old man with trigeminal neuralgia
over left V2 and V3 dermatomes. He had a previous transarterial
embolization and long use of carbamazepine with partial response
to treatment. Gamma knife radiosurgery (GKR) was planned. A
marginal dose of 15 Gy was given to 50% isodose line. His pain
was relieved by GKR in 1.5 years. Treatment of posterior fossa
arteriovenous malformations causing trigeminal neuralgia, with
GKR has a very limited use in the literature. It, however, is obvious
that success rate as pain relief, in a very challenging field of
functional neurosurgery, is satisfactory. Large series, however,
are in need to make a more comprehensive statement about efficacy
and safety of the procedure in these pathologies.
Key Words: Arteriovenous malformation, embolization, Gamma
knife radiosurgery, trigeminal neuralgia
FIGURE 1. Gamma knife radiosurgery plan for the cerebellopontine angle

arteriovenous malformation.
rigeminal neuralgia (TN) is a facial pain syndrome characterized as sudden onset and lightening-like sensation over somatosensorial branch(es) of fifth cranial nerve (trigeminal nerve).
Trigeminal nerve has 3 branches: ophthalmic (V1), maxillary (V2),
and mandibular (V3). The distribution of pain over both V2 and V3
dermatomes is usual pattern of the syndrome.1 Rarely, some underlying diseases or disorders could be diagnosed, such as multiple
sclerosis, brain tumors, and vascular malformations. Trigeminal
neuralgia caused by cerebellopontine arteriovenous malformation
(AVM) is very rare. We discuss the case and therapeutic approach of
Gamma knife radiosurgery (GKR) with a review of literature.
CLINICAL REPORT
A 47-year-old man admitted to our Gamma knife unit in October 2013
with left-sided facial pain, headache, memory problems, and difficulty
in articulation. The pain was excruciating and had an electric shocklike character distributed over his left maxilla and mandibula. In his
past medical history, he had had a transarterial embolization treatment
for a cerebellopontine angle AVM in 1995. The pain had persisted
despite the embolization procedure. He had used carbamazepine for a
long period with some extent of relief. His quality of life and
functional capabilities had become limited in time. In his neurologic
examination, there was no neurologic deficit except a mild hypoesthesia over the left V2 and V3 dermatomes. On brain magnetic resonance,
a left cerebellopontine AVM was observed. Gamma knife radiosurgery was planned for the AVM. A marginal dose of 15 Gy was
given to 50% isodose line (Fig. 1). The pain resolved in a stepwise
manner during his 1.5 years of follow-up (Fig. 2).
From the Department of Neurosurgery, Hakkari State Hospital, Hakkari,
Turkey; yDepartment of Orthopaedic Surgery-Spine Center, University
of California at San Francisco, San Francisco, CA; and zDepartment of
Neurosurgery, Bahcesehir University Medical School, Istanbul, Turkey.
Received August 21, 2015.
Address correspondence and reprint requests to Murat Sakir Eksi, MD,
Department of Orthopaedic Surgery, University of California at San
Francisco, 500 Parnassus Avenue MU320 West, San Francisco, CA
941430728; E-mail: muratsakireksi@gmail.com
Murat Sakir Eksi, MD, was supported by a grant from TUBITAK (The
Scientific and Technological Research Council of Turkey), Grant
number: 1059B191400255.
ISSN: 1049-2275
DOI: 10.1097/SCS.0000000000002310
e56
FIGURE 2. Follow-up at 1 year (A) and 1.5 years (B).
DISCUSSION
Trigeminal neuralgia is a pain syndrome expressed as electric shock
like, stabbing pain appearing in episodes. Chewing, swallowing,
brushing teeth, speaking, and touching the face may trigger the pain
episodes. Main pathophysiological event behind this pain syndrome
is compression of trigeminal nerve by a vessel (most probably a
branch of superior or anterior inferior cerebellar artery) at or near
the root entry zone of the nerve, which leads to demyelination and/
or ectopic action potential production of the nerve.2,3 Prolonged
after-discharge at the trigger zone has been proven with neurophysiological monitoring of the trigeminal nerve.4
Incidence of trigeminal neuralgia has been reported as
4.7/100,000.5 Some underlying diseases, such as multiple sclerosis,
brain tumors, and AVMs were defined as the causative factors.
Surgery, stereotactic radiosurgery, and embolization have been
conveyed as treatment tools for cerebellar AVMs causing TN.6
Hemorrhagic complications of AVM surgery that is performed
after embolization procedure has been reported as 6% in Spetzler
Martin Grade III and as 20% to 25% in SpetzlerMartin Grade IV
and Grade V AVMs. Even though success rate of GKR in AVMs
after embolization is somewhat lower than sole GKR (41% versus
59%), complication rates are similar.7 Because embolization procedure had already been used and did not work out, we planned
GKR for the cerebellopontine angle AVM.
Gamma knife radiosurgery has been used in the previous 7 cases
that harbored posterior fossa AVM causing TN (Table 1).3,6,812
Including our case, mean and median ages of patients were 53.75
and 52.5 years, respectively (range 3969 years). There is a
male predominance (male:female ratio is 7:1). Anderson et al9 and
#
TABLE 1. Posterior Fossa Arteriovenous Malformations and Dural Arteriovenous Fistulas, Causing Trigeminal Neuralgia, Treated With Gamma Knife Radiosurgery
Procedure
Author(s)/Year
Age/Sex
Other Additional
Interventional Treatment
Sato et al/2003
49/F
MVD after GKR
Pain relief 1 year after the surgery
Karibe et al/2004
Anderson et al/2006
Matsushige et al/2006
Lesley/2009
55/M
39/M
50/M
55/M
MVD before GKR

None
None
Embolization before GKR
Sumioka et al/2011
66/M
MVD before GKR
Mori et al/2014
Present case
69/M
47/M

Pain relief after MVD

Pain relief 6 months after GKR
Pain relief 1 year after GKR
Pain relief after 4 months of embolization, GKR
applied for residual AVM
Pain relief after surgery, nidus disappearance 18
months after GKR
Pain relief 1.5 years after GKR
Pain relief 1.5 years after GKR
Outcome
Complications
Pain increased gradually
1 year after GKR
None
None
None
None
None
None
None
F, female; GKR, Gamma knife radiosurgery; M, male; MVD, microvascular decompression.
Matsushige et al12 reported successful use of sole GKR for TN because

of AVM. Other combinations to GKR, before or after, were microvascular decompression surgery of trigeminal nerve or embolization
of the AVM. Of 8 patients, GKR primarily cured TN in 4 patients,
GKR was used as an adjuvant therapy to eradicate or take under control
the AVMs in 3 patients.3,6,912 Median time to pain relief in 4 patients
was 15 months (range 618 months).3,9,12 In 1 case, reported by
Sato et al8, TN could not be relieved by GKR, instead was treated with
microvascular decompression of the trigeminal nerve.
CONCLUSIONS
Treatment of posterior fossa AVMs causing TN, with GKR has a
very limited use in the literature. It, however, is obvious that success
rate as pain relief, in a very challenging field of functional neurosurgery, is satisfactory. Large series, however, are in need to make a
more comprehensive statement about efficacy and safety of the
procedure in these pathologies.
ACKNOWLEDGMENT
The authors thank Selim Olduz for his technical support during
manuscript preparation.
REFERENCES
1. Zakrzewska JM, Linskey ME. Trigeminal neuralgia. BMJ Clin Evid
2014;2014:1207
2. Eller JL, Raslan AM, Burchiel KJ. Trigeminal neuralgia: definition and
classification. Neurosurg Focus 2005;18:E3
3. Mori Y, Kobayashi T, Miyachi S, et al. Trigeminal neuralgia caused by
nerve compression by dilated superior cerebellar artery associated with
cerebellar arteriovenous malformation: case report. Neurol Med Chir
(Tokyo) 2014;54:236241
4. Burchiel KJ, Baumann TK. Pathophysiology of trigeminal neuralgia: new
evidence from a trigeminal ganglion intraoperative microneurographic
recording. Case report. J Neurosurg 2004;101:872873
5. Katusic S, Williams DB, Beard CM, et al. Epidemiology and clinical
features of idiopathic trigeminal neuralgia and glossopharyngeal
neuralgia: similarities and differences, Rochester, Minnesota, 1945
1984. Neuroepidemiology 1991;10:276281
6. Lesley WS. Resolution of trigeminal neuralgia following cerebellar
AVM embolization with Onyx. Cephalalgia 2009;29:980985
7. Xu F, Zhong J, Ray A, et al. Stereotactic radiosurgery with and
without embolization for intracranial arteriovenous malformations:
a systematic review and meta-analysis. Neurosurg Focus 2014;37:E16
8. Sato K, Jokura H, Shirane R, et al. Trigeminal neuralgia associated with
contralateral cerebellar arteriovenous malformation. Case illustration.
J Neurosurg 2003;98:1318
#
9. Anderson WS, Wang PP, Rigamonti D. Case of microarteriovenous

malformation-induced trigeminal neuralgia treated with radiosurgery.
J Headache Pain 2006;7:217221
10. Sumioka S, Kondo A, Tanabe H, et al. Intrinsic arteriovenous
malformation embedded in the trigeminal nerve of a patient with
trigeminal neuralgia. Neurol Med Chir (Tokyo) 2011;51:639641
11. Karibe H, Shirane R, Jokura H, et al. Intrinsic arteriovenous
malformation of the trigeminal nerve in a patient with trigeminal
neuralgia: case report. Neurosurgery 2004;55:1433
12. Matsushige T, Nakaoka M, Ohta K, et al. Tentorial dural arteriovenous
malformation manifesting as trigeminal neuralgia treated by stereotactic
radiosurgery: a case report. Surg Neurol 2006;66:519523
Primary W-Plasty Closure for

Surgical Repair of the Injured Lip
Ali Gomez, DDS and Alexander Pomares, DDSy
Purpose: To describe the results obtained using the primary Wplasty closure technique for the surgical repair of lip defects of
traumatic etiology.
Patients and Methods: This study followed both the Declaration of
Helsinki on medical protocol and ethics and the Ethical Guidelines of
Hospital San Francisco de Asis institutional review board. A retrospective case series study was designed and implemented. The medical
status, demographic, etiology, complications, and outcomes associated to the primary W-plasty technique in patients with lip injuries seen
at Hospital San Francisco de Asis Oral and Maxillofacial Surgery
Department in Quibdo, Colombia between 2010 and 2013 were
assessed. Data were collected and analyzed using a statistic package.
Results: Eighteen patients were treated, 8 women and 10 men, with
ages ranging from 2 to 38 years. Patients presented lip avulsion
From the Oral and Maxillofacial Surgery Department; and yGeneral Dentistry Department, Hospital San Francisco de Asis, Quibdo, Colombia.
Received August 21, 2015.
Address correspondence and reprint requests to Ali Gomez, DDS, Oral and
Maxillofacial Surgery Department, Hospital San Francisco de Asis, Calle
31 #5-43 piso 1, Quibdo, Choco, Colombia;
E-mail: gomezren@hotmail.com
ISSN: 1049-2275
DOI: 10.1097/SCS.0000000000002311
e57
TABLE 1. Summary of Patients With W-Plasty Identified at Hospital San Francisco de Asis Oral and Maxillofacial Surgery Department From 2010 to 2013
Variables
FIGURE 1. Basic W-plasty technique used in all patients. A, Clinical

presentation. B, Wound debridement. C, Flap design. D, Closure.
FIGURE 2. This 32-year-old man presented with a traumatic lesion involving

more than 50% of the lower lip. The injury, which was caused by a human bite,
comprised mucosa, semimucosa, and the vermilion border. A, Clinical
presentation. B, Mucocutaneous flap design. C, At 1-day follow-up. D, Clinical
presentation after 1 week of evolution.
injuries associated to different etiologies. They underwent surgery

under general anesthesia after prophylactic antibiotic therapy and
tetanus booster vaccination. Defect repair was performed using the
primary W-plasty closure technique, obtaining satisfactory esthetic
and functional results.
Conclusions: Primary W-plasty closure is a surgical option that
allows the safe and practical restoration of the injured lip, even in
lesions exceeding 50% of the normal lip.
Result
Patients (N 18)
Men
Women
Involved Lip
Upper
Lower
Etiology
(1) Facial trauma
Men
Women
(2) Multiple trauma
Men
Women
(3) Lip-only trauma
Man
Woman
(4) Human bite
Man
Woman
(5) Gunshot wound
Man
Woman
(6) Short blunt weapon wound
Male
Woman
(7) Posttraumatic macrostoma
Man
Woman
Surgical Technique
W-plasty
Complications
Intraoperatively
Postoperatively
Infection and necrosis
Outcomes
Adequate esthetics
Adequate lip mobility
Sensitivity
Hypertrophic scar
10 (55.6%)
8 (44.4)
0
18 (100%)
7 (38.88%)
4
3
4 (22.22%)
2
2
2 (11.11%)
1
1
2 (11.11%)
1
1
1 (5.55%)
1
0
1 (5.55%)
1
0
1 (5.55%)
1
0
18 (100%)
0
1 (5.55%)
1
18
18
18
6
lip injuries seen at Hospital San Francisco de Asis in Quibdo,

Colombia between 2010 and 2013. This study followed the Declaration of Helsinki on medical protocol and ethics.
A total of 18 patients, 10 men (56.60%) and 8 women (44.40%)
were treated. The samples average age was 24.9 years (range
238). The surgical technique consisted of a W-shaped myocutaneous flap, lip remodeling, resection of avulsed necrotic tissues,
and progressive reconstruction of the muscle, mucosa, and skin
(Figs. 1 and 2). Table 1 summarizes the main findings. All patients
received antibiotic and tetanus therapy.
ACKNOWLEDGMENT
Key Words: Lip reconstruction, W-plasty, facial trauma
ip reconstruction is a complex procedure, because the reproduction of esthetic lip using limited amounts of tissue may lead to
complications related to function and self-esteem.1 An increase in
the frequency of lip injuries has been documented, with the great
majority of cases requiring surgical reconstruction.2 Currently,
there is a paucity of articles documenting the applications of
W-plasty approach for lip reconstruction. The purpose of this article
is to present the outcomes associated to this method in patients with
e58
The authors thank Jaime Castro-Nunez and Glenna Castro for

their help.
REFERENCES
1. Fernandez R, Clemow J. Outcomes of total or near-total lip
reconstruction with microvascular tissue transfer. J Oral Maxillofac
Surg 2012;70:28992906
2. Cavalcanti AL, Bezerra PKM. Maxillofacial injuries and dental
trauma in patients aged 1980 years. Rev Esp Cir Oral Maxilofac
2010;32:1116
Rigid External Distractor Aided

Conventional Le Fort III
Osteotomy Advancement in
Adult With Severe
Midfacial Hypoplasia
Ruichen Wang, MDy and Chunming Liu, MD, DDSy
Background: Management of severe midfacial hypoplasia is still a
challenge for craniofacial team, adult patients with syndromic midfacial hypoplasia made the situation even worse. The authors present
the clinical result in an adult patient with Crouzon syndrome treated
by rigid external distractor aided conventional Le Fort III procedure.
Methods: A 26-year-old patient with Crouzon syndrome presented
with severe midfacial hypoplasia, a negative overjet of 17 mm,
exorbitism, airway obstruction, and masticatory problem, while
chief complaint of the patient was abnormal appearance. After Le
Fort III osteotomy, rigid external distractor and distraction hooks
were fixed. With the aid of rigid external distractor, the midfacial
mass was immediately advanced to a desired position, allograft
bone grafted in the gaps, and microplate fixed. The device was
removed 3 weeks later when the advanced midface was stable.
Results: Point Awas advancedby18.6 mminhorizontaland displaced
superiorly 0.5 mm in vertical at the time of device removal. Point A
moved backward 1.5 mm and upward 0.3 mm at 2-year follow-up. A
good and balanced facial profile was obtained in a short treatment
period. Airway obstruction symptoms and exorbitism were relieved.
Conclusions: The midfacial advancement achieved by rigid external
distractor aided conventional Le Fort III osteotomy is a stable, controllable, and timesaving procedure. This technique may become an
important choice for adult patients with severe midfacial hypoplasia.
Key Words: Adult, Crouzon syndrome, Le Fort III osteotomy,
severe midfacial hypoplasia
evere midfacial hypoplasia is a typical skeletal deformity in

syndromic craniosynostosis, such as Apert, Crouzon, and Pfeiffer syndrome. These syndromes may present with variable deformities, but share one common characteristic that the midfacial
hypoplasia caused by premature fusion of sutures is a three-dimensional skeletal defect involving maxilla, nasal complex, and zygomatic body.1,2 Clinical features may include exorbitism, Class III
skeletal malocclusion, upper airway obstruction, and facial dysmorphism.3 Treatment aims at correction of midface hypoplasia as
well as related functional, morphologic, and psychologic problems.
From the Department of Burns and Plastic Surgery, Chinese PLA 309
Hospital; and yDepartment of Plastic and Reconstructive Surgery,
Chinese PLA Medical School, Beijing, China.
Address correspondence and reprint requests to Chunming Liu, MD, DDS,
Department of Plastic and Reconstructive Surgery, Chinese PLA
Medical School & General Hospital, 28 Fuxing Road, Beijing 100853,
China; E-mail: liuchm301@163.com
ISSN: 1049-2275
DOI: 10.1097/SCS.0000000000002232
#
Syndromic midfacial hypoplasia are usually corrected with surgical advancement on Le Fort III level. Surgical procedure can be
performed by using either conventional Le Fort III osteotomy or Le
Fort III distraction osteogenesis (DO) technique.4 Since a large amount
of advancement is required in patients with severe midfacial hypoplasia, DO is preferred over conventional procedure.
Distraction osteogenesis has been proven to achieve 2- to 3-fold
advancement than conventional procedure with a lower relapse
rate.5,6 Although there are no clinical guidelines regarding the ideal
timing and planning of these surgical procedures, it is generally
believed that DO of the Le Fort III level should be performed at an
age-appropriate time to maximize the effects of normal skeletal
growth and reduce the need for further surgical intervention.
Patients before skeletal maturity have clearly benefited from DO
procedure.1 Patients after skeletal maturity are generally recommended conventional orthognathic surgery for its short treatment
period and high patient compliance.4,6,7 Conventional procedure,
however, commonly could not achieve enough advancement or
provide sufficient stability in severe midfacial hypoplasia.
The combination of Le Fort III and Le Fort I osteotomy seemed
to be applied in patients after skeletal maturity.4,7 The combining
osteotomy may achieve good result on the occlusion, but compromises on upper portion of the midface. Therefore, it may produce an
inharmonious facial appearance appeared a protruded mouth and
retro-positioned suborbit, nose and zygoma. Treatment of adult with
severe midfacial hypoplasia is technically demanding.
In this study, we present our experience of combining conventional Le Fort III osteotomy and rigid external device in the
treatment of an adult patient with Crouzon syndrome.

Patients
A 26-year-old woman with Crouzon syndrome presented with
severe midfacial hypoplasia, exorbitism, masticatory problem, and
upper airway obstruction, while the chief complaint was abnormal
appearance (Fig. 1). Clinical examination revealed a concave profile
and a class III molar relationship, a negative overjet of 17 mm.
The cephalometric radiographs were obtained at 3 time intervals:
preoperation (T1), device removal (T2), and 2-year follow-up (T3).
Preoperative lateral cephalogram indicated severe midfacial hypoplasia and slight mandible hyperplasia (Fig. 2). Three-dimensional computed tomographic examinations (SIEMENS Sensation 16, Germany)
were performed before surgery and 3-month follow-up (Fig. 3).
Cephalometric Analysis
Traditional cephalometric measurements were taken. The lateral
cephalometric radiographs at the 3 periods were superimposed on
the anterior cranial base, and the differences in point A and point B
on the x- and y-axis were calculated. The x-axis was parallel to the
Frankfort plane, and the y-axis was normal to x-axis at Sella (S) in
this coordinate system.
Surgical Procedure
Complete subperiosteal exposure of the interested areas through
lower eyelid, intraoral, and upper palpebral incisions. A modified
Le Fort III osteotomy was performed through naso-frontal suture,
the medial and inferior orbital wall, the zygomatic body, pterygomaxillary and septal disjunction. After the separation of midfacial
mass, the distraction hooks were fixed on the each side of nasal floor
and introduced from the nostril.
After the fixation of rigid external distractor system, double
25-gauge stainless steel wires were used to connect the distraction
e59
FIGURE 1. A 26-year-old woman with Crouzon syndrome: A, preoperative

frontal view; B, preoperative right view; C, frontal view at 3 months
postoperatively; and D, right view at 3 months postoperatively.
FIGURE 3. Preoperative and postoperative computed tomography scans: A,

preoperative front views of skeleton; B, preoperative lateral views of skeleton
(yellow, oblique) and soft tissue (blue, semitransparency); C, postoperative front
views of skeleton; and D, preoperative lateral views of skeleton and soft tissue.
with her facial appearance. The mandible hyperplasia and anterior

open bite were not corrected because the scheduled bilateral sagittal
split osteotomy of the mandible did not proceed.
Cephalometric Results
FIGURE 2. Serial lateral cehpalograms: A, before Surgery; B, device removal;

and C, 2-year follow-up.
hooks to the distraction screws on the cross-arms of the vertical bar.

The midfacial mass was advanced by rotating clockwise the activation screws. As the Le Fort III osteotomy mass reached at a desired
position, 5 pieces of allograft bone were grafted in the advancement
defects. The midface mass was stabilized with rigid fixation.
Postoperative treatment
After a 3-week period of consolidation, when the activation
screws were rotated anticlockwise gradually, there was no drawbacks. The device was removed in outpatient.
Traditional cephalometric analysis data are shown in Table 1.

Point A was advanced by 18.6 mm along the x-axis and displaced
superiorly by 0.5 mm along the y-axis between T1 and T2 period.
Point B was advanced by 2.5 and 4.8 mm, respectively. Point A
showed posterior movement of 1.5 mm on the x-axis and upward
movement of 0.3 mm on the y-axis between T2 and T3 period. Point
B showed anterior and upward movement of 0.4 and 2.0 mm,
respectively (Figs. 2 and 4).
DISCUSSION
Traditionally, patients with syndromic midfacial hypoplasia were
treated with orthognathic surgery, which involves a Le Fort III
osteotomy advancement and rigid fixation with or without bone
grafts. In severe cases, it is, however, not only technically difficult
to advance the deficient skeleton to the normal position, but also has
a high rate of relapse.8 The introduction of DO makes the correction
of severe midfacial hypoplasia easier, safer, and more efficacious.9
RESULTS
TABLE 1. Traditional Lateral Cephalometric Measurements at 3 Time Points
Clinical Results
Patient had obvious improvement in facial convexity and
appearance. A good balanced facial profile was obtained (Figs. 1
and 3). Airway obstructive symptoms and exorbitism were relieved,
and masticatory function was improved. Intraoral examination
showed an acceptable molar relationship. The patient was satisfied
e60
Measurement
T1
T2
T3
SNA, degrees
SNB, degrees
ANB, degrees
65.5
84.5
18.2
82.7
84.8
2.2
81.1
84.5
4.0
T2-T1
T3-T2
17.1
0.4
15.9
1.6
0.4
1.8
FIGURE 4. Seriallateralcephalometric superimposition ofthepatient preoperation

(T1), device removal (T2), and 2-year follow-up (T3) shown the advancement of
midfacial mass and the stability of advancement during the 2 stages.
Distraction osteogenesis has been approved to achieve greater midfacial skeletal advancements with a lower relapse rate compared with
conventional procedure.6,10 As DO technique refined, Le Fort III DO
has become an important choice, particularly in the treatment of young
patients with severe midfacial retrusion or airway obstruction.11
At the time of this writing, the best treatment of syndromic
midfacial hypoplasia is to stage reconstruction coincided with facial
growth patterns. Some patients from remote rural in China, however, have not accessed the treatment until the completion of
skeletal maturity, even if they appeared obvious deficiency at early
period of adolescent. Therefore, these patients still suffered from
extremely severe deformities after grownup and the treatment
became more complicated and uncontrollable.
The application of DO in adult is limited by the long period of
treatment, high rate of relapse, and unpredictable results of
advancement.11,12 Because the duration of consolidation phase
depends on the severity and age of patients, adult patients or large
advancing may require long period of duration.13,14 Achieving large
advancement of midface is crucial to the treatment of severe cases;
however, there is obvious resistance to the midfacial advancement
appeared at the late of active period. Furthermore, there are higher
rate of relapse in adults, mainly at first 6 months postoperatively.
Former studies suggested that 20% overcorrection in midfacial
advancement was required in adult patients.8 Therefore, further
orthognathic surgery in midface will probably be required for a
desired outcome after the use of distraction technique.14
It is noted that a dual-distraction technique combined with the
use of external and internal distractors was applied to maintain the
efficacy and stability.1416 This technique, however, took at least a
3-month period of consolidation and required a second intervention
to the removal of internal distractor. Entire treatment period is so
long that this technique may be impracticable in adults. As a result,
the application of DO in this challenge group of patients requires a
long period of treatment, but still might lead uncertainty results and
beyond the surgeons control.4,7
Owing to the restriction of soft tissue to the bony advancement,
conventional Le Fort III procedure is technically difficult to achieve
large advancement.4,6 In addition, immediate advancement beyond
10 mm would indicate high rate of relapse. Le Fort III with an
additional Le Fort I osteotomy was suggested in adult patients,
and the results varied from positive or good to excellent.4,7 The
combining osteotomy may achieve more advancement at the occlusal
level. Patients who present unequal severity of retrusion at the orbital
and the occlusal levels, and need more advancement at occlusal level,
may benefit from this procedure, but it is incomplete correction in
syndromic midfacial hypoplasia. In addition, the combined osteotomy increased the complexity of surgical procedure.12
In this study, the severe hypoplasia midfacial mass was advanced
immediately under the aid of rigid external distractor. The device
plays 2 roles: the first is to overwhelm the resistance of soft tissue and
make the midface mass advanced largely and precisely to its anatomy
#
position; the second is to stable the midface mass in its advanced

position and resist the withdrawal force of soft tissue until the soft
tissue stretched and gradually adapted to new skeletal position. With
the aid of distractor, it is not only technically simple and precise to
advance the midface to a desired position, but also relatively controllable to maintain the midface position to prevent relapse.
In syndromic midfacial hypoplasia cases, the distractor is generally kept in place at least for 10 weeks after active distraction
period,17 and on our experience the devices were kept at least
12 weeks in adult patients. Compared with previous cases, this
technique has achieved as great advancement as DO, whereas the
entire treatment period was shortened to 3 weeks. With respect to
stability, cephalometric analysis of 2-year follow-up showed there
was 1.5-mm orizontal relapse (8.6%) and minimal changes
(0.3 mm) in vertical. The costs were increased compared with
conventional Le Fort III procedure; however, only in pins and
traction hooks of distractor which are definitively single use, the
halo, distractor arm and vertical bar can be reused.12 Compared with
a repeated Le Fort III procedure, the costs were not increased.
In this study, the authors aimed to achieve an aesthetic and stable
result in a relative short treatment period. The mandible hyperplasia
and final occlusal relation were scheduled at 6 months later when
the advanced midface was stable. The scheduled operation in
mandible, however, could not proceed because the patient was
satisfied with the appearance outcome and postoperative occlusion.
CONCLUSIONS
The application of distractor added extra step of fixation of distraction, but did not increase the complexity of procedure and
actually made the conventional Le Fort III procedure technically
easier in skeletal advancement and fixation, and preventing of
relapse became controllable. This technique could achieve large
advancement with a stable and predictable aesthetic outcome in a
short treatment period. The rigid external device aided conventional
Le Fort III osteotomy minimize the possibility of additional
orthognathic surgery in midface and provide an alternative choice
in the treatment of adult patients with severe midfacial hypoplasia.
ACKNOWLEDGMENTS
The authors thank Dr Yusheng Yao for collecting follow-up data.
Yusheng Yao, Department of Stomatology, the Third Affiliated
Hospital of Liaoning Medical University.
REFERENCES
1. Iannetti G, Ramieri V, Pagnoni M, et al. Le Fort III external midface
distraction: surgical outcomes and skeletal stability. J Craniofac Surg
2012;3:896900
2. Rice DP. Clinical features of syndromic craniosynostosis. Front Oral
Biol 2008;12:91106
3. Phillips JH, George AK, Tompson B. Le Fort III osteotomy or
distraction osteogenesis imperfecta: your choice. Plast Reconstr Surg
2006;4:12551260
4. Nout E, Cesteleyn LL, van der Wal KG, et al. Advancement of the
midface, from conventional Le Fort III osteotomy to Le Fort III
distraction: review of the literature. Int J Oral Maxillofac Surg
2008;9:781789
5. Fearon JA. The Le Fort III osteotomy: to distract or not to distract? Plast
6. Saltaji H, Altalibi M, Major MP, et al. Le Fort III distraction
osteogenesis versus conventional Le Fort III osteotomy in correction
of syndromic midfacial hypoplasia: a systematic review. J Oral
7. Nout E, Koudstaal MJ, Wolvius EB, et al. Additional orthognathic
surgery following Le Fort III and monobloc advancement. Int J Oral
e61
8. Liu C, Liu C, Gao Q, et al. Transsutural distraction osteogenesis versus

osteotemy distraction osteogenesis. J Craniofac Surg 2012;2:464471
9. Liu C, Hou M, Liang L, et al. Sutural distraction osteogenesis (SDO)
versus osteotomy distraction osteogenesis (ODO) for midfacial
advancement: a new technique and primary clinical report. J Craniofac
Surg 2005;4:537548
10. Caterson EJ, Shetye PR, Grayson BH, et al. Surgical management of
patients with a history of early Le Fort III advancement after they
have attained skeletal maturity. Plast Reconstr Surg 2013;4:
592e601e
11. Heggie AA, Kumar R, Shand JM. The role of distraction osteogenesis in
the management of craniofacial syndromes. Ann Maxillofac Surg
2013;1:410
12. William H, Bell, Guerrero CA. Distraction Osteogenesis of the Facial
Skeleton. Hamilton, Ontario: BC Decker Inc; 2007:269-298
13. Lee DW, Ham KW, Kwon SM, et al. Dual midfacial distraction
osteogenesis for Crouzon syndrome: long-term follow-up study
for relapse and growth. J Oral Maxillofac Surg 2012;3:
e242e251
14. SantAnna EF, Cury-Saramago Ade A, Lau GW, et al. Treatment of
midfacial hypoplasia in syndromic and cleft lip and palate patients by
means of a rigid external distractor (RED). Dental Press J Orthod
2013;4:134143
15. Lee DW, Ham KW, Kwon SM, et al. Dual midfacial distraction
osteogenesis for Crouzon syndrome: long-term follow-up study for
relapse and growth. J Oral Maxillofac Surg 2012;3:e242251
16. Figueroa AA, Polley JW. Management of the severe cleft and syndromic
midface hypoplasia. Orthod Craniofacial Res 2007:167179
17. Nada RM, Sugar AW, Wijdeveld MG, et al. Current practice of
distraction osteogenesis for craniofacial anomalies in Europe: a web
based survey. J Craniomaxillofac Surg 2010;2:8389
Is Radiologic Evaluation
Necessary to Find out Foreign
Bodies in Nasal Cavity?
Hoon Oh, MD, Hyun Jin Min, MD, PhD,
Hoon Shik Yang, MD, PhD, and Kyung Soo Kim, MD, PhD
Abstract: Although there were previous studies on the clinical
aspects such as etiology, treatment modalities, studies regarding the
necessity of radiologic evaluation for nasal foreign body were
limited. The aim of this study is to evaluate the necessity and
indication of radiologic evaluation for nasal foreign bodies. There
are consecutive patients aged less than 10 years who presented with
suspected foreign bodies in nasal cavity. We reviewed the patients
age and sex, including the methods of evaluation, management
tools, and types of foreign bodies. There were 35 cases (11.4%) on
whom radiographs were performed in the 24 uncooperative patients
and 11 cooperative patients who were not identified with any
From the Chung-Ang University College of Medicine, Seoul, South Korea.
Accepted for publication August 13, 2015.
Address correspondence and reprint requests to Kyung Soo Kim, MD, PhD,
Department of Otorhinolaryngology-Head and Neck Surgery, ChungAng University College of Medicine, 224-1, Heukseok-dong, Dongjak-gu,
Seoul 56-755, South Korea; E-mail: 9921045@hanmail.net
ISSN: 1049-2275
DOI: 10.1097/SCS.0000000000002165
e62
foreign bodies via nasal endoscopy. Among them, only 4 cases

had positive reports of foreign body and the others were normal
radiologic findings. We suggest that the radiologic evaluation is
always not necessary to find the location of nasal foreign bodies. It,
however, should be performed in cases of negative findings of
physical examination with anterior rhinoscopy or sinus endoscopy
and unwitnessed foreign bodies to rule out metallic contents,
especially button type battery.
Key Words: Evaluation, foreign body, nasal cavity, radiology
he etiology of nasal foreign bodies has been ascribed to general

curiosities and whims to explore nasal orifices in children,
playful insertion of foreign bodies into others body parts,
accidental entry of foreign body, habitual cleaning of nose.1 Nasal
foreign bodies can be categorized as organic/inorganic, animate/
inanimate, metallic/nonmetallic, hygroscopic/nonhygroscopic,
regular/irregular, and soft/hard according to their nature.2 A nasal
foreign body usually can be easily removed because endoscopic
evaluation is accessible.3 Delayed treatment has been correlated
with more severe lesions, in addition to more complications.4 There
have been reported many ways to remove foreign bodies in nasal
cavity, and the choice of removal modality usually depends on the
type of foreign body, its position, and patients cooperation. Also,
the rate of success relies on the location and characteristics of
foreign bodies, the physicians experience, the equipment available,
and patients cooperation.3
Although there have been many previous studies on the clinical
aspects such as etiology and management modality, studies regarding radiologic evaluation of nasal foreign body are few. So, the aim
of this study is to evaluate the necessity of radiologic studies.

A retrospective study was done on consecutive patients aged less
than 10 years old, who presented with suspected nasal foreign
bodies in our hospital over a 9-year period between January 2005
and March 2014. We reviewed the patients age and sex, including
the evaluation methods, management modality, and types of foreign
bodies. We performed sino-nasal endoscopy in all patients who
were cooperative, and simple radiologic studies were performed if
patients were not. Hospitalization was generally required when
general anesthesia was necessary to proceed with the removal of the
foreign body.
RESULTS
There were 307 cases of nasal foreign body, with 147 men (48%)
and 160 women (52%). The majority of the patients were less than
3 years old (81.3%) and the mean age was 2.6 years (range from
2 months to 7 years) (Fig. 1). Our study shows 192 patients (62.5%)
presented with right-sided nasal foreign body, 107 patients (34.9%)
in the left side, and 8 patients (2.6%) in both nasal cavity. The most
FIGURE 1. Age distribution.
FIGURE 2. The type of nasal foreign body.
common type of nasal foreign bodies was round-shaped materials,

such as balls, marbles, and beads which were consisted of
54 patients (17.6%). This was followed by plastic toys in 51 patients
(16.6%), nuts and seeds (such as peas, beans) in 51 patients (16.6%),
and papers in 28 patients (9.1%) (Fig. 2).
When the patient was presented with nasal foreign body,
we removed visible foreign bodies through only anterior
rhinoscopy in cooperative patients at emergency department
(111 patients, 36.2%). If nasal foreign body, however, was not
removed by the emergency department, the patients would be
immediately referred to the ear nose and throat (ENT) department (196 patients, 63.8%). Overall, 146 patients (47.2%) were
able to remove foreign bodies through ENT intervention with
nasal endoscopy. There, however, were 50 patients (16.2%) who
we could not find anything.
Most foreign bodies in nose were directly removed by forceps,
hooks, and suctioning under sinus endoscopy. There were 4 uncooperative patients (1.4%) who underwent removal procedure under
general anesthesia and they have been hospitalized for 1 day on
an average. There were 35 patients (11.4%) on whom radiologic
evaluations were performed: there were 24 uncooperative patients
and 11 cooperative patients who were not identified with any
foreign bodies with sinus endoscopy but were supposed to have
metallic materials (magnets, batteries, etc). Only 4 patients had
positive reports of foreign body and the others were normal radiologic findings.
DISCUSSION
The age of patients who insert foreign bodies into nasal cavity is
less compared with the age of patients who present with other
ENT problems. This trend is also noted in previous reports.5 7
The majority of the patients were less than 3 years old (81.3%).
Young children are curious and will insert foreign bodies into
their noses, usually with objects found at home.8 In our study,
balls and marbles are the most common materials of nasal foreign
bodies, closely followed by toys and nuts. These are the objects
readily available and reachable by the children in most of the
houses. Interestingly, we found that right nasal cavity was more
commonly affected (60.0%) than left nasal cavity (34.9%), and
we suppose that predominance of right-handedness might be the
reason for this.
In general, direct visualization with anterior rhinoscopy is
usually enough to identify and locate nasal foreign bodies. If
the nasal foreign body is not seen or witnessed, sinus endoscopy
for viewing the entire nasal cavities may be used to identify the
location.3
Although some authors suggest that radiologic evaluation
should only be performed in patients suspected with foreign
bodies, when careful physical examination and nasal endoscopy
failed to produce additional evidence,9 there is no definite indication. In this study, we performed radiographs when a patients
#
FIGURE 3. A, X-ray shows a radio-opaque foreign body (asterisk) is between

nasal septum and right inferior turbinate. B, Sino-nasal endoscopy shows a nasal
foreign body supposed as metal is in the same location. C, Normal radiograph.
D, Sino-nasal endoscopy shows a nasal foreign body with purulent discharge is
between nasal septum and right inferior turbinate. Likewise, radiologic imaging
is not useful in the diagnosis of radiolucent objects.
foreign body was uncooperative, unwitnessed, and unfound via

sinus endoscopy. In this study, 35 patients had to undergo radiologic
study such as paranasal sinus series, and 4 patients (11.4%) had
positive findings: circular magnet (2 patients) and button type
battery (2 patients). Radio-opaque foreign bodies, such as metal,
glass, and stone foreign bodies could be clearly found with simple
x-rays.10 The frontal and lateral views with a double-contour feature
are useful to identify the button type battery9 (Fig. 3A-B). Simple
x-ray, however, is not useful in the identification of radiolucent
objects. (Fig. 3C-D)
Although most of nasal foreign bodies are relatively safe, easily
detected and promptly removed in the outpatient clinic or emergency room, some cases such as button type battery may require
more complicated ways such as general anesthesia and special
instruments.11 Button type battery may cause severe complications
such as adhesion of nasal cavity, perforation of the nasal septum,
and saddle nose.12 The removal methods of nasal foreign body are
various, including direct removal with the use of various instruments, the use of Fogarty biliary catheter, and the mothers kiss
(parents kiss) technique.13,14
In our study, emergency department physicians could successfully manage most children presented with nasal foreign bodies.
The goal of management, however, should be to minimize complications and morbidity, which often result from repeated attempts
for removal. If emergency department physicians cannot find a
foreign body with anterior rhinoscopy, they should refer to ENT
department. Emergency department physicians give patients the
information of complications induced by excessive procedure for
removal and having foreign body in the nasal cavity for over
24 hours. Also, they should explain that some foreign bodies, such
as button type battery, are needed to be removed as soon as possible
and are useful to find the content and location through radiologic
evaluations (Fig. 4).
e63
Obturator Prostheses for

Melanotic Neuroectodermal
Tumor of Infancy in the Maxilla
Thas Bianca Brandao, MSc,
Aljomar Jose Vechiato-Filho, MSc,
and Alan Roger dos Santos Silva, PhDy
FIGURE 4. Algorithm of approach to the nasal cavity foreign body in pediatric

patients.
CONCLUSIONS
Our study suggests that the radiologic evaluation is not necessary to
find the location of nasal foreign bodies. It, however, should be
performed in cases of negative findings of physical examination
with anterior rhinoscopy or nasal endoscopy and unwitnessed
foreign bodies to rule out metallic contents, especially button type
battery.
REFERENCES
1. Kalan A, Tariq M. Foreign bodies in the nasal cavities: a comprehensive
review of the aetiology, diagnostic pointers, and therapeutic measures.
Postgrad Med J 2000;76:484487
2. Schulze SL, Kerschner J, Beste D. Pediatric external auditory canal
foreign bodies: a review of 698 cases. Otolaryngol Head Neck Surg
2002;127:7378
3. Heim SW, Maughan KL. Foreign bodies in the ear, nose, and throat. Am
Fam Physician 2007;76:11851189
4. Tiago RS, Salgado DC, Correa JP, et al. Foreign body in ear, nose and
oropharynx: experience from a tertiary hospital. Braz J Otorhinolaryngol
2006;72:177181
5. Baker MD. Foreign bodies of the ears and nose in childhood. Pediatr
Eemer Care 1987;3:6770
6. Nandapalan V, McIlwain JC. Removal of nasal foreign bodies with
a Fogarty biliary balloon catheter. J Laryngol Otol 1994;108:
7587607
7. Kadish HA, Corneli HM. Removal of nasal foreign bodies in the
pediatric population. Am J Emerg Med 1997;15:5456
8. Balbani AP, Sanchez TG, Butguan O, et al. Ear and nose foreign
body removal in children. Int J Pediatr Otohinolaryngol 1998;46:
3742
9. Lin VY, Daniel SJ, Papsin BC. Button batteries in the ear, nose and
upper aerodigestive tract. Int J Pediatr Otohinolaryngol 2004;68:
473479
10. Aras MH, Miloglu O, Barutcugil C, et al. Comparison of the sensitivity
for detecting foreign bodies among conventional plain radiography,
computed tomography and ultrasonography. Dentomaxillofac Radiol
2010;39:727813
11. Yasny JS. Nasal foreign bodies in children: considerations for the
anesthesiologist. Paediatr Anaesth 2011;21:11001102
12. Thabet MH, Basha WM, Askar S. Button battery foreign bodies in
children: hazards, management, and recommendations. Biomed Res Int
2013;2013:846091
13. Fox JR. Fogarty catheter removal of nasal foreign bodies. Ann Emerg
Med 1980;9:3738
14. Glasziou P, Bennett J, Greenberg P, et al. Mothers kiss for nasal foreign
bodies. Aust Fam Physician 2013;42:288289
e64
Abstract: Melanotic neuroectodermal tumor of infancy frequently

affects the maxilla. A communication between the oral and nasal
cavities can be created by surgery. The authors rehabilitated a
young patient with obturator prostheses to correct feeding. The
association of the obturators with orthodontic devices provided
proper maxillary growth and eruption of teeth. The outcomes were
very satisfactory after a 3-year follow-up, and dental implants are
planned.
Key Words: Case reports, melanotic neuroectodermal tumor,
palatal obturators
elanotic neuroectodermal tumor of infancy (MNTI) is a rare

lesion that affects the head and neck region (68%78% at the
anterior region of maxilla) of children during their first year of
life.1 4 Approximately 7% of MNTI tumors are malignant, with 3
patients reported in the maxilla, and show a similar histological
pattern with increased mitosis, hypercellularity, and focal necrosis.5
There are a limited number of articles on maxillary MNTI.
We searched the PubMed database using the following strategy:
(neuroectodermal tumor, melanotic [MeSH Terms] OR
(neuroectodermal [All Fields] AND tumor [All Fields]
AND melanotic [All Fields]) OR melanotic neuroectodermal
tumor [All Fields] OR (melanotic [All Fields] AND neuroectodermal [All Fields] AND tumor [All Fields] AND infancy[All Fields]) OR melanotic neuroectodermal tumor of
infancy[All Fields]). Only 301 articles were found. Thus, the
purpose of this brief case report is to provide information as to
how obturator prostheses might be a useful tool to restore maxillary
defects created by surgery and facial aesthetics. To our knowledge,
this is the first report of an obturator for an MNTI patient.
CLINICAL REPORT
A 7-month-old boy was referred to the Hospital of the Faculty of
Medicine of Sao Paulo University with a painless swelling tumor
located in the anterior region of the alveolar ridge of the maxilla. An
intraoral examination revealed a lobed and ill-defined sessile bluish
From the Dental Oncology Service, Instituto do Cancer do Estado de Sao
Paulo (ICESP), Faculdade de Medicina da Universidade de Sao Paulo,
Sao Paulo; and yOral Diagnosis Department, Piracicaba Dental School,
University of Campinas (UNICAMP), Piracicaba, Sao Paulo, Brazil.
Address correspondence and reprint requests to Thas Bianca Brandao, MSc,
Instituto do Cancer do Estado de Sao Paulo (ICESP), Dr. Arnaldo
Avenue, 251, Cerqueira Cesar, Sao Paulo, Sao Paulo 01246-000, Brazil;
E-mail: thais.brandao@icesp.org.br
ISSN: 1049-2275
DOI: 10.1097/SCS.0000000000002228
CONCLUSION
FIGURE 1. A, Oronasal communication created by ablative surgery. B,

Obturator prosthesis associated with an orthodontic device for the eruption
of teeth.
mass (2 cm) at the right anterior maxillary region (from the right
primary canine to the left primary lateral incisor). The area was
scanned by computerized tomography, revealing an extensive illdefined, homogeneous, hypo-dense tumor associated with bone
resorption and displacement of the unerupted tooth germs. Routine
laboratory test results were within normal limits, including levels of
urinary vanillylmandelic acid (UVA) below 6 mg. This is a rare
situation because increases of UVA levels are generally associated
with MNTI diagnosis.4 An incisional biopsy was performed for
histopathological diagnosis, which was conclusive for MNTI.3
Impressions of the maxilla and mandible were made with
irreversible hydrocolloid (Jeltrate, Dentsply, Petropolis, RJ, Brazil) before surgery. The surgical procedure included resection of
the anterior maxilla (including the nasal floor) and excision of the
tumor with safe margins (5 mm margin all around). An acrylicbased healing plate was fabricated for immediate surgical obturation to allow proper feeding. The healing plate was inserted and
adjusted with a soft liner (CoeSoft; GC America, Alsip, IL), then
removed 3 weeks after surgery, and replaced with an obturator
prosthesis. When the patient reached 2 years, the orthodontic
treatment was associated to facilitate tooth eruption (Fig. 1).
The patients recovery from surgery was unremarkable and no
signs of lesion recurrence were observed after 3 years of follow-up.
Several changes of the obturator prostheses were necessary to
compensate for maxillary growth over time. In addition, an orthopedic device was attached to the obturator prosthesis to improve
occlusion relationships, provide normal speech, and improve facial
aesthetics. Prosthetic rehabilitation with osteo-integrated dental
implants is planned for the patient after the complete development of
his maxilla.
DISCUSSION
The etiology of MNTI is unknown, and surgery is the primary
treatment.1,6 A 5 mm resection margin was used when removing the
tumor, as previously described.1,5 8 This wide excision margin is
recommended for MNTI since there is a chance of recurrence.5
Rustagi et al8 observed that 2.53% of maxillary MNTI tumors were
malignant in 237 patients, but Kruse-Losler et al5 affirm this rate
may range from 10% to 45%. In our patient, no recurrence was
observed during a 3-year follow-up.
Since most MNTI tumors occur in the maxilla (68%78%),1 4
functional and/or aesthetic defects might result from surgery because
a partial or complete maxillectomy is necessary for tumor
removal.1,6,7 To the best of our knowledge, there are no reports of
patients with MNTI who were treated with obturator prostheses. In
our patient, an oronasal communication was created during surgery
and a prosthodontist and an orthodontist tried to correct the defect.
The obturators we used enabled a proper deglutition and eliminated the need for a nasogastric tube. In addition, the association of
the obturator with the orthodontic devices did not interfere with
maxillary growth or teeth eruption. The management of a patient
with MNTI should involve a multidisciplinary team whenever
possible since the surgery might remove teeth germs and affect
maxillary growth. Moreover, the outcome of a further prosthetic
rehabilitation might be compromised. In our patient, the use of
obturator prostheses allowed the planning of dental implants.
#
A dental examination is recommended for the treatment of MNTI.

In addition, a long-term follow-up is mandatory. Obturator prostheses should be encouraged for MNTI with communications
because the prostheses enables deglutition, eliminates the need
for a nasogastric tube, enables maxillary growth, and, consequently,
provides a better scenario for further prosthetic rehabilitation.
REFERENCES
1. Gupta R, Gupta R, Kumar S, et al. Melanotic neuroectodermal tumor of
infancy: review of literature, report of a case and follow up at 7 years.
J Plast Reconstr Aesthet Surg 2015;68:5354
2. Selim H, Shaheen S, Barakat K, et al. Melanotic neuroectodermal tumor
of infancy: review of literature and case report. J Pediatr Surg
2008;43:2529
3. Tabrizi R, Bahramnejhad E, Kazemi H, et al. Rapidly growing lesions
involving the maxilla in infants: a two-case presentation and deferential
diagnosis. J Craniofac Surg 2013;24:434438
4. Kruse-Losler B, Gaertner C, Burger H, et al. Melanotic neuroectodermal
tumor of infancy: systematic review of the literature and presentation of a
case. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2006;102:
204216
5. Kruse-Losler B, Gaertner C, Burger H, et al. Melanotic neuroectodermal
tumor of infancy: systematic review of the literature and presentation
of a case. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2006;102:
204216
6. Beogo R, Nikiema Z, Traore SS, et al. Maxillary melanotic
neuroectodermal tumor of infancy management: is conservative
surgery the best approach? J Craniofac Surg 2013;24:e338e340
7. Rachidi S, Sood AJ, Patel KG, et al. Melanotic neuroectodermal tumor of
infancy: a systematic review. J Oral Maxillofac Surg 2015;73:19461956
8. Rustagi A, Roychoudhury A, Karak AK. Melanotic neuroectodermal
tumor of infancy of the maxilla: a case report with review of literature.
J Oral Maxillofac Surg 2011;69:11201124
Cranial Fasciitis: A Systematic

Review and Diagnostic
Approach to a Pediatric
Scalp Mass
Ryan D. Wagner, BS, Eric K. Wang, BS,
Mark S. Lloyd, MD, Sandi K. Lam, MD,y
and David Y. Khechoyan, MD
Abstract: Cranial fasciitis is an uncommon, benign fibroproliferative condition of the scalp or skull that arises in children. Clinically,
it manifests as a firm, nontender, subcutaneous, enlarging mass.
From the Department of Surgery, Division of Plastic Surgery; and
yDepartment of Surgery, Division of Neurosurgery, Baylor College of
Medicine, Houston, TX.
Address correspondence and reprint requests to David Y. Khechoyan, MD,
6701 Fannin St. Suite 610.00, Houston, TX 77030;
E-mail: David.khechoyan@bcm.edu
ISSN: 1049-2275
DOI: 10.1097/SCS.0000000000002230
e65
The purpose of our study was to review the literature on cranial

fasciitis to create a diagnostic algorithm using the latest patient at
our institution as an example. The authors conducted a systematic
review examining all published cases of cranial fasciitis in English
literature. The authors then created a diagnostic algorithm to help
distinguish cranial fasciitis from other similarly presenting cranial
masses. To demonstrate this algorithm, the authors detailed the
latest patient with cranial fasciitis at our institution. The authors
extracted data from 53 published reports documenting 72 patients of
cranial fasciitis. Our patient presented similarly to what was
reported in the literature. A 7-week-old boy presented with 2 small
parietal scalp masses that were noted shortly after birth. After
noncontrast computed tomography imaging, the enlarging masses
were resected and found to have eroded the outer cranial vault
cortex. Histological analysis revealed cranial fasciitis. The differential diagnosis for an enlarging scalp mass in an infant or child is
broad. Cranial fasciitis cannot be diagnosed based on clinical
presentation alone. Imaging is usually employed to further characterize lesions after initial examination but histopathological
analysis is essential for diagnosis. The locally invasive nature of
cranial fasciitis makes it difficult to distinguish from malignant
conditions such as sarcomas. However, if the diagnosis of cranial
fasciitis is considered early, patients can achieve prompt clinical
resolution following simple resection.
Key Words: Cranial fasciitis, differential diagnosis, scalp mass
ranial fasciitis is a benign fibroproliferative condition of the

pediatric scalp that lies along the spectrum of diagnoses
associated with an enlarging scalp mass. To date, 72 patients have
been reported in the English literature since its original description
in 1980.1 Although variable, patients typically present with a firm,
nonpainful, and rapidly enlarging scalp lesion.1 8 Lesions are
predominantly localized to the subcutaneous tissue or galea aponeurotica. However, awareness of cranial fasciitis is important due
to a transcranial invasive potential.3,5,7 10 These lesions may occur
anywhere on the cranium but are most frequently reported on the
temporal and parietal regions.3,5,8,9
The extensive differential on initial presentation of an enlarging
scalp mass in a pediatric patient can pose a diagnostic difficulty
for clinicians. By systematically reviewing all patients of cranial
fasciitis previously reported in the literature, we were able to
compare the clinical characteristics of cranial fasciitis to other
pediatric scalp lesions and create a diagnostic algorithm that can
aid clinicians in the diagnostic process.
METHODS
Literature Review
A systematic literature search was conducted through PubMed
and Scopus using Cranial Fasciitis, Nodular Fasciitis AND
Skull, and Nodular Fasciitis AND Cranium as the search
criteria. Reported patients with a diagnosis of cranial fasciitis or
nodular fasciitis of the cranium fulfilled our inclusion criteria.
Poster presentations, conference abstracts, and non-English articles
were excluded.
Data extracted from eligible articles included the number of
patients with cranial fasciitis, age of presentation, sex, history of
previous head trauma, anatomical site, radiological imaging
modality, depth of invasion, presence of ossification or calcification, recurrence after excision, and follow-up time for postoperative
e66
surveillance. Not all studies reported on all of the above variables.

Descriptive statistics were used to quantify the data extracted from
studies that fulfilled our inclusion criteria.
As part of our ongoing institutional case series for cranial
fasciitis, our most recent case was used to highlight the characteristic features of the presentation and the application of our
diagnostic algorithm.
RESULTS
Literature Review
Since first described by Lauer and Enzinger in 1980, there have
been 53 published reports documenting 72 patients of cranial
fasciitis in the English literature.1 Patients had a median age at
presentation of 2 years, with a male-to-female ratio of roughly 1.5 to
1 (44:28). The most frequently encountered anatomical sites of
occurrence were the temporal (39%) and parietal (25%) regions.
Cranial fasciitis occurred less frequently in the occipital and frontal
regions. Four patients with exclusive intracranial involvement have
been reported.1114 These masses were all extra-axial and appeared
to arise from the dura.
Of the 49 patients with information on trauma history,
14 reported prior trauma to the cranium at the location of the
cranial fasciitis (29%).1,5,7,10,1522 An additional 4 patients reported
cranial fasciitis that occurred within a region of prior radiation
therapy.5,7,23,24
The imaging modality most often used to characterize the scalp/
skull masses was computed tomography (CT) scan of the head,
followed by MRI of the brain, with many patients undergoing both
imaging investigations. Plain film x-ray and ultrasound were used
with less frequency. In 11 patients, ossification of the soft tissue
mass was observed (Table 1).1,7,19,2527
Recurrence of the lesion following excision was uncommon and
reported in only 3 cases in the literature.21,28,29 However, 18 patients
were lost to follow-up, so the true rate of recurrence may be
underestimated due to lack of long-term monitoring. Based on
the 54 patients of cranial fasciitis with documented follow-up, a
recurrence rate of 5.7% after excision was calculated. Of the
3 patients failing initial treatment, 1 patient displayed recurrence
3 weeks after resection of the lesion but, after repeat excision, the
patient was reported to be lesion-free at 9 months.28 Information
regarding the secondary treatment for the other 2 patients of
recurrence was not reported.
Clinical Report
The most recent patient with cranial fasciitis at our institution is
described highlighting the presentation, clinical course, and treatment of cranial fasciitis. A 7-week-old adopted boy born at 38 weeks
gestational age presented with 2 small parietal scalp masses that
were noted shortly after birth. The masses were painless and seemed
stable in size from birth. There was limited prenatal care and in
utero exposure to alcohol and tobacco. The details of labor and
delivery were not known.
Physical examination revealed 2 well-circumscribed 1 cm hard,
rubbery lesions 3 cm left of the midline in the parietal region. The
lesions were mobile with the scalp. There was no skin discoloration,
erythema, or tenderness to palpation. The remainder of the examination was benign. The lesions evolved on serial examination. By 4
months of age, the masses had grown in size and were adherent to
the skull. A noncontrast CT scan of the head showed minor
subcutaneous soft tissue thickening overlying the high left parietal
region with the report offering the potential diagnosis of a resolving
cephalhematoma (Fig. 1).
#
TABLE 1. Cases of Cranial Fasciitis Published in the English Literature From 1980 to October 2014
Author
Year
Age (yr)
Sex
Site of Lesion
Curtin E30
Lecavalier M8
Fissenden TM31
Hattab EM14
Kong WK32
Wu B23
Yiu Y27
Garza L33
Liu CC34
Imafuku S35
du Toit LE36
Marshall LR37
Johnson KK7
2014
2014
2014
2014
2014
2013
2013
2012
2011
2011
2009
2009
2008
Rakheja D29
2008
Hussein MR10
Takeda N38
SantaCruz K39
Yebenes M6
Clark M26
Summers LE5
2008
2008
2007
2007
2007
2007
Oh CK40
Agozzino M13
Kim ST15
Lee JY16
Longatti P24
Keyserling HF3
Larralde M4
Rapana A12
Govender PV41
Pollack IF42
Marciano S9
Sajben FP28
Skoog L43
Martinez-Lage JF44
Boddie DE17
Clapp CG45
Hoya K18
Lang DA46
Pagenstecher A11
Sayama T47
Iqbal K48
Hoeffel JC49
Hunter NS50
Sato Y19
Kumon Y51
Inamura T20
Coates DB52
Mollejo M25
Patterson JW2
2007
2006
2005
2004
2004
2003
2003
2002
2001
2001
1999
1999
1999
1997
1997
1997
1996
1996
1995
1995
1995
1993
1993
1993
1992
1991
1990
1990
1989
Adler R21y
Ringsted J53
1986
1985
2
4
1.1
2.7
1.5
13
1.5
2
0.6
5
0.1
0.2
0.3
11
0.3
0.3
1
1
0.3
5.3
2.2
3.3
2
3
27
8
2.5
16
61
0.3
8
22
3
11
0.6
0.2
47
2
0.1
34
1.5
2
6
2.5
0.1
1.2
0.3
7
0.8
5
6
0.8
7
5
2
3
11
7
3
1.7
6
F
M
M
F
F
F
F
F
F
M
M
M
M
F
M
M
M
F
F
F
M
M
M
F
F
M
M
F
M
M
F
F
F
M
M
M
M
M
F
M
M
F
M
F
M
M
M
F
M
F
M
M
M
M
M
M
F
M
F
M
M
Lt Temporal
Rt Temporal
Rt Petrous Temporal
Lt Frontoparietal
Rt Maxilla
Rt Occipital
Rt Maxilla
Rt Temporoparietal
Rt Frontoparietal
Lt Frontal
Rt Frontal
Rt Petrous Temporal
Rt Frontoparietal
Rt Parietal
Mid Occipital
Mid Occipital
Mid Occipital
Rt Temporoccipital
Rt Temporoparietal
Lt Occipital
Rt Orbital
Lt Occipital
Mid Occipital
Rt Temporal
Rt Temporal
Occipital
Lt Occipital
Parasagittal
Rt Temporoparietal
Lt Temporal
Lt Frontotemporal
Mid Frontoparietal
Rt Petrous Temporal
Lt Temporal
Rt Parietal
Temporal
Bregma
Lt Parietal
Lt Petrous Temporal
Rt Frontal
Rt Orbital
Rt Frontoparietal
Lt Frontotemporal
Rt Petrous Temporal
Lt Temporal
Rt Temporal
Rt Temporal
Lt Frontal
Rt Occipital
Lt Forehead
Mid Occipital
Rt Temporoparietal
Lt Temporal; Rt Temporoparietal
Lt Parietal
Trauma
Imaging
No
No
No, Radiation
No
No
No
No
No
No
No
Yes
No, Radiation
Yes
No
No
No
Yes, Radiation
Yes
Yes
Yes
No, Radiation
No
No
No
No
Yes
No
Yes
No
No
No
No
Yes
Yes
No
No
No
Yes; Yes
CT
CT
CT and MRI
CT and MRI
CT
CT and MRI
CT
CT and MRI
CT and MRI
US and CT
CT and MRI
CT
CT and MRI
CT and MRI
CT and MRI
CT and MRI
US, CT, MRI

CT and MRI
MRI
X-Ray
CT and MRI
MRI
CT and MRI
CT and MRI
CT and MRI
CT
CT
MRI
CT and MRI
US and MRI
CT and MRI
CT and MRI
CT and MRI
CT and MRI
MRI
US and CT
X-Ray
CT and MRI
CT and MRI
CT and MRI
CT and MRI
CT and MRI
CT
US and CT
CT
US and CT
CT and MRI
CT
CT
CT
X-ray
X-ray
Depth of Invasion
Dura
Dura
Cranium
Dura
Cranium
Dura
Cranium
Dura
Dura
Galea
Subcutaneous
Cranium
Dura
Dura
Periosteum
Extracranialunspecified
Dura
Cranium
Subcutaneous
Dura
Dura
Cranium
Dura
Subcutaneous
Subcutaneous
Dura
Dura
Subcutaneous
Dura
Dura
Dura
Cranium
Periosteum
Dura
Cranium
Dura
Dura
Dura
Dura
Dura
Dura
Cranium
Cranium
Dura
Subcutaneous
Periosteum
Dura
Extracranial unspecified
Cranium; Cranium
Dura
e67
TABLE 1. (Continued)
Author
22
Barohn RJ
Lauer DH1
Year
Age (yr)
Sex
1980
1980
1
1.8
6
0.2
5
0.1
1.5
0.3
1.5
0.6
F
M
F
M
F
M
F
M
M
M
Site of Lesion
Trauma
Lt Parietal
Rt Temporal
Mid Occipital
Rt Parietal
Rt Temporal
Mid Frontal
Temporoparietal
Lt Temporal
Rt Frontoparietal
Occipitoparietal
Imaging
Yes
CT
Yes (1)
No (8)
X-ray
Depth of Invasion
Cranium
Dura (3)
Extracranialunspecified (5)
(1)
X-ray was only recorded when it was the sole imaging modality utilized.
y
Author reported on a single patient with 2 temporally distinct patients with cranial fasciitis.
By 8 months of age, the 2 scalp lesions were fixed and contiguous, measuring 2.5 cm by 1.2 cm. The overlying skin had
developed grey discoloration. The patient was recommended to
undergo excisional biopsy given the change in character and growth
of the lesion. On surgical exposure, the lesion tightly adhered to the
galea aponeurotica superiorly with bony erosion of the outer cranial
vault cortex. Thorough curettage of the outer cortex showed no
FIGURE 1. Noncontrast computed tomography scan of the head in a 3-mo-old

with an enlarging, firm, nontender scalp mass. Sagittal (A) and coronal (B) views
show minor subcutaneous soft tissue swelling with slight hyperintensity overlying
the high left parietal region. No cranial involvement is noted at this time.
FIGURE 2. Characteristic histopathological features of cranial fasciitis in a 2.5 cm

1.2 cm scalp mass excised from an 8-mo-old patient. Examination shows
proliferation of spindled fibroblasts and myofibroblasts imbedded in a collagenous
and myxoid stroma. Scattered lymphocytes and vascular proliferation with
extravasated red blood cells are seen throughout (A, hematoxylin and eosin stain
40). Features of nuclear atypia and prominent mitotic figures are absent (B,
hematoxylin and eosin stain 200). Immunohistochemistry displays diffusely
positive SMA staining (C, smooth muscle actin 200) and a cytoplasmic pattern of
staining with beta-catenin (D, beta-catenin 200). For comparison, images B, C,
and D display the same region of tissue.
e68
transcranial invasion. The inner cortex remained intact after the

mass was removed.
Histological analysis revealed a fibroblastic and myofibroblastic
proliferation with spindle-shaped cells, arranged in a storiform
pattern with scattered lymphocytes. Nuclei were bland-appearing
without significant atypia or increased mitotic figures. Areas of
dense connective tissue with low cellularity were also present.
Smooth muscle actin (SMA) stain was diffusely positive and beta
catenin stain displayed a cytoplasmic pattern consistent with cranial
fasciitis (Fig. 2).
At 2-week postoperative follow-up, the patients wound was
healing well. The patients family reported the appearance of a
small nodular mass at the lateral edge of the previous resection
6 weeks after surgery, which persisted at recent 3-month follow-up.
This lesion is suspicious for local recurrence and will undergo
workup and re-excision.
DISCUSSION
The differential diagnosis for an enlarging scalp mass in an infant or
child is broad. Cranial fasciitis cannot be diagnosed based on
clinical presentation alone. Imaging and biopsy are useful tools
to distinguish cranial fasciitis from similarly presenting conditions.
The clinical findings in our case report of an infant with a firm,
nontender, subcutaneous, and enlarging mass in the parietal
region were consistent with cases previously described in the
literature.1 4,6 8 The underlying etiology of cranial fasciitis is
uncertain. A relationship with cranial trauma has previously been
proposed,3,5,710 though patients have been reported in the absence
of previous trauma and also after radiation therapy.
Imaging is usually employed to further characterize lesions after
initial examination. CT scan of the head is nonspecific but can
provide information regarding underlying skeletal involvement.6
Lytic lesions are observed when the tumor has eroded into the
FIGURE 3. A diagnostic algorithm to aid in the diagnosis of cranial fasciitis given

the differential of an enlarging scalp mass in a pediatric patient. Similarly
presenting conditions can be systematically ruled-out (R/o) based on clinical
presentation, imaging results, and microscopic findings of biopsy specimens.
cranium.1,3,7 Sclerosis and ossification of the lesion may or may not

be present.3,5,6 MRI of the brain in cranial fasciitis displays more
unique features for diagnosis. T1-weighted imaging typically
demonstrates hypointensity, vivid enhancement, and a nonenhancing central region. T2-weighted imaging shows hyperintensity in
the central nonenhancing area.3,5,7
Histopathologic examination is essential for the diagnosis of

cranial fasciitis. Histologically, cranial fasciitis mimics nodular
fasciitis, which characteristically presents as a benign solitary soft
tissue nodule in the subcutaneous tissue, fascia, or muscle of the
upper extremities in individuals 20 to 35 years of age.1,2,5,6 Cranial
fasciitis is a proliferative lesion of myofibroblasts and fibroblasts
TABLE 2. Differential Diagnosis for a Pediatric Patient Presenting With an Enlarging Scalp Mass
Clinical Presentation
Differential Diagnosis
H&P - > R/o
Infantile
Hemangioma
Pilar cyst
Pilomatricoma
History
Rapid growth followed by

slow involution
Slow-growing mass normally
on scalp, often multiple
Solitary slow growing mass
Caput succedaneum
Swelling arising after birth

and resolving within days
Cephalohematoma
Swelling after prolonged

labor or instrument use,
resolving within weeks
H&P - > Image - > R/o

Encephalocele
Eosinophilic
Granuloma
Leptomeningeal cyst
Spherical bluish midline

mass, compressible, may
transilluminate
Usually a single region of

bone pain and swelling
Focal tnderness, swelling,

softening of underlying
bone
Soft mass at site of previous

head trauma
Painful palpable mass, back

pain, systemic symptoms
Microscopic Examination
Soft cystic mass, nontender,

often pulsatile
Variable, cranial nerve

defects
Tender mass overlaying a

bone, petechiae,
neuropathic pain
US: cranial defect, cystic,

brain defect
MRI: superior detail
XR: lytic lesions
CT: detects intracranial

extension
XR: sharply marginated linear
lucency
CT: fracture, dural tear
XR: majority radiolucent
CT: determines intracranial
extension
XR: lytic lesion, onion skin
periosteal reaction
Variable, necessary for

diagnosis
Small round blue cells
CT & MRI: tumor extent

Osteosarcoma
Dermoid Cyst
Dermatofibrosarcoma
Protuberans
Infantile
Myofibromatosis
Pain with activity, often in

knee shoulder or hip,
swelling, prior trauma
Additional Tests
Cutaneous: blanching red

well-circumscribed
SubQ: blue rubbery tense
Smooth nodule, mobile, wellcircumscribed, skin colored
Hard subcutaneous mass, skin
colored to bluish,
ulceration
Soft tissue swelling crosses
suture lines, poorly defined
margins
Subperiosteal swelling
restricted by suture lines,
well defined margins
Soft mass, can enlarge

with crying
H&P - > Image - > Biopsy - > R/o

benign skull tumor
Variable, enlarging skull
mass, pain
Ewing Sarcoma
Physical Examination
Biopsy
Imaging
X-Ray or CT or MRI
Tender often warm

erythematous mass,
decreased ROM
Variable, slow growing mass

with tuft of hair, unilateral
eyelid swelling
Slow growing skin mass
Cystic mass or cutaneous

sinus, often around eyes,
nontender
Skin colored to reddish
plaque composed of
nodules, ulceration
Usually a single nodule

present at birth or
developing shortly after
Skin-colored to purple firm

nodule involving skin or
muscle, non-tender
XR: periosteal elevation
CT & MRI: tumor location

and extent
MRI: detects intracranial cyst,
differentiates from other
cysts
CT: bone involvement or
metastasis
MRI: tumor border, depth
US: anechoic center
MRI: low signal on

T1-weighted imaging
Immunostain:
CD99
Molecular:
t(11;22)
Presence of osteoid, spindle

shaped stromal cells
Mature epidermal
appendages, hair follicles,
sebaceous glands
Spindle tumor cells in a
storiform pattern, often no
defined boarder
Spindle cells, collagen, areas
of necrosis and increased
vascularity
Immunostain:
CD34
Immunostain:
vimentin
and smooth
muscle actin
CT, computed tomography; H&P, history and physical; R/o, rule-out; US, ultrasound; XR, x-ray.
e69
embedded in a collagenous and myxoid stroma. The cells are

spindle or stellate shaped and organized in a storiform or nodular
pattern. Vascular proliferation with extravasation of red blood cells,
inflammatory cell infiltration, and multinucleated giant cells are
often observed.1,2,4,6 Metaplastic ossification and matrix calcification can be seen.6,9 As expected for a fibroplastic proliferative
process, immunohistochemistry reveals positive staining for vimentin and SMA.2,5 7,9 Microscopic examination of the biopsy from
our patient showed the characteristic histological features outlined
above; no reactive bone formation was observed. An immunohistochemical stain for beta-catenin localized to the cytoplasm, which
is consistent with the staining pattern of nodular fasciitis.29
The standard treatment for cranial fasciitis is resection.1 3,8,10
An incisional biopsy is necessary to rule out a malignant process
and to confirm a diagnosis of cranial fasciitis. If the lesion is small
and the scalp wound can be closed directly, an excisional biopsy
may be performed, which would alleviate the need for a second
operation. Recurrence following excision is uncommon, but may be
under-reported. Barring surgical resection, the use of intralesional
corticosteroids is a potential medical treatment if the overall clinical
picture is highly consistent with cranial fasciitis. One case report
described the use of intralesional triamcinolone acetonide injected
twice with a 10-day interval and no recurrence after 4-month
follow-up.16
A differential diagnosis should include other conditions that
produce an enlarging scalp mass in a pediatric patient. The following diagnoses should also be considered upon initial presentation:
infantile hemangioma, pilar cyst, pilomatrixoma, caput succedaneum, cephalohematoma, encephalocele, eosinophilic granuloma,
leptomeningeal cyst, benign skull tumors, Ewing sarcoma, osteosarcoma, dermoid cyst, dermatofibrosarcoma protuberans (DFSP),
and infantile myofibromatosis.13,5,6,8,9 To reach the diagnosis of
cranial fasciitis, these conditions can be systematically ruled out
after a thorough history and physical examination, appropriate
imaging, and histopathological examination of specimens
(Fig. 3). Table 2 outlines the characteristic clinical presentation,
imaging results, and histopathological findings for the above-listed
conditions to aid in the differentiation from cranial fasciitis
(Table 2). Other lesions to consider in the differential diagnosis
include meningioma, osteomyelitis, traumatic lesions, sarcoidosis,
surface metastases, and syphilis.2,3,5,6,9
Cranial fasciitis is an uncommon condition which should be
included in the differential diagnosis of an infant or young child
presenting with an enlarging scalp/skull mass. The clinical course
characterized by a rapidly enlarging lesion and the locally invasive
nature of cranial fasciitis can make it difficult to distinguish from
malignant conditions such as sarcomas. If the diagnosis of cranial
fasciitis is considered early, patients can achieve prompt clinical
resolution following a simple resection.
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Anthropometric Analysis
of the Face
Georgios V. Zacharopoulos, MD,y Andreas Manios, MD, PhD,y
Chung H. Kau, MScD, PhD,z George Velagrakis, MD, PhD,
George N. Tzanakakis, MD, PhD,jj and Eelco de Bree, MD, PhDy
Background: Facial anthropometric analysis is essential for planning cosmetic and reconstructive facial surgery, but has not been
available in detail for modern Greeks. In this study, multiple
measurements of the face were performed on young Greek males
and females to provide a complete facial anthropometric profile
of this population and to compare its facial morphology with that of
North American Caucasians.
Materials and Methods: Thirty-one direct facial anthropometric
measurements were obtained from 152 Greek students. Moreover,
the prevalence of the various face types was determined. The
#
resulting data were compared with those published regarding North

American Caucasians.
Results: A complete set of average anthropometric data was
obtained for each sex. Greek males, when compared to Greek
females, were found to have statistically significantly longer foreheads as well as greater values in morphologic face height, mandible width, maxillary surface arc distance, and mandibular surface
arc distance. In both sexes, the most common face types were
mesoprosop, leptoprosop, and hyperleptoprosop. Greek males had
significantly wider faces and mandibles than the North American
Caucasian males, whereas Greek females had only significantly
wider mandibles than their North American counterparts.
Conclusions: Differences of statistical significance were noted in
the head and face regions among sexes as well as among Greek and
North American Caucasians. With the establishment of facial norms
for Greek adults, this study contributes to the preoperative planning
as well as postoperative evaluation of Greek patients that are,
respectively, scheduled for or are to be subjected to facial reconstructive and aesthetic surgery.
Key Words: anthropometry, facial anthropometry, Greeks, North
American Caucasians
uantitative evaluation of facial morphology by using anthropometric measurements is essential for surgeons when planning facial surgery, either as reconstructive surgery after trauma and
oncological resections or as an aesthetic procedure. At the time of
this writing, many disciplines such as pediatrics, medical genetics,
orthodontics, and craniofacial surgery commonly use anthropometric measurements of the face in clinical assessment, diagnosis,
and reconstruction planning. Furthermore, facial anthropometric
values have been used to study the differences among various ethnic
or racial groups.14 The facial morphology elucidates variations
among populations of different ethnic background. Using a single
standard for all ethnic groups is not appropriate as it can lead to
aesthetically unsatisfactory results. Therefore, specific facial norms
should be available for each ethnic group. Unfortunately, such data
are lacking for many ethnic groups and the differences among them
have been insufficiently studied.
The Ancient Greeks were the first to perform measurements of the
human face. The Greek sculptor Polycleitus in the 5th century BC
described the ideal proportions of the human face,5 and introduced
canons that evolved to the neoclassical canons of the face and which
From the Technological Educational Institute of Crete, Heraklion, Greece;
yDepartment of Surgical Oncology, Medical School of Crete University
Hospital, Heraklion, Greece; zDepartment of Orthodontics, University of
Alabama at Birmingham, Birmingham, AL; Department of Otorhinolaryngology, Medical School of Crete University Hospital, Heraklion,
Greece; and jjLaboratory of Anatomy-Histology-Embryology, Medicinal School of Crete, Heraklion, Greece.
Received April 20, 2015.
Address correspondence and reprint requests to Georgios V. Zacharopoulos,
MD, Department of Surgical Oncology, Medical School of Crete
University Hospital, P.O. Box 1352, 71110 Heraklion, Greece;
E-mail: gzachar@hotmail.com
Supplemental digital contents are available for this article. Direct URL citations
appear in the printed text and are provided in the HTML and PDF versions
of this article on the journals Web site (www.jcraniofacialsurgery.com).
ISSN: 1049-2275
DOI: 10.1097/SCS.0000000000002231
e71
are still being used today as a road map for facial operations. In a
previous study, we, however, demonstrated that in the majority of
modern Greeks the neoclassical facial canons are not valid.6
Several studies have established normative data of facial
measurements in North American Caucasians, African Americans,1
Koreans,7 and other ethnic groups.2,3 Although the Greeks first
performed measurements of the human face, normative values of
the Greek face have been insufficiently determined. In a multicenter
study,4 the late professor Leslie Gabriel Farkas (19152008), who
is considered the pioneer of modern facial anthropometry, and coworkers examined the craniofacial characteristics of 25 ethnic
groups including Greeks. In this study, a limited number (14) of
parameters, however, were determined in a relatively small number
of Greek participants (30 males and 30 females).
The aim of the current study is to establish a complete normative
anthropometric facial profile of the modern Greek population, based
on considerably more parameters in a much larger cohort. In addition,
these anthropometric facial norms were compared with those of North
American Caucasians to determine whether there are significant
differences in facial characteristics between these populations.

Patients
A total of 152 volunteer students, 78 men and 74 women, from
the Technological Educational Institute of Crete were studied. The
principles outlined in the Declaration of Helsinki were followed and
informed consent was obtained. All students and all their 4 grandparents were of Greek origin. These volunteers had normal body
mass index, no history of reconstructive or aesthetic operations to
the face and no apparent facial anomalies. The mean age of the
volunteers was 22.5 years (range 1830).
A large group of North American Caucasian males and females
who had participated in a previous study4 was used as reference in
the search for significant facial differences among North American
Caucasians and the studied Greek population.
TABLE 1. Analyzed Anthropometric Landmarks (Farkas et al9)

Anthropometric Landmarks
Gonion (go)
The most lateral point on the mandibular angle close to the
bony gonion
Gnathion (gn)
The lowest median landmark on the lower border of the
mandible
Trichion (tr)
The point on the hairline in the midline of the forehead
Nasion (n)
The point in the midline of both the nasal root and the
nasofrontal suture
Glabella (g)
The most prominent midline point between the eyebrows
Opisthocranion (op)
The point situated in the occipital region of the head and is
most distant from the glabella
Zygion (zy)
The most lateral point of each zygomatic arch
Endocanthion (en)
The point at the inner commissure of the eye fissure.
Exocanthion (ex)
The point at the outer commissure of the eye fissure
Alare (al)
The most lateral point on each alar contour
Subnasale (sn)
The midpoint of the columella base at the apex of the angle
where the lower border of the nasal septum and the
surface of the upper lip meet
Subalare (sbal)
The point at the lower limit of each alar base, where the alar
base disappears into the skin of the upper lip
Crista philtri
The point on each elevated margin of the philtrum just above
landmark (cph)
the vermilion line
Labiale (or labrale)
The midpoint of the upper vermilion line
superius (ls)
Labiale (or labrale)
The midpoint of the lower vermilion line
inferius (li)
Stomion (sto)
The imaginary point at the crossing of the vertical facial
midline and the horizontal labial fissure between gently
closed lips, with teeth shut in natural position
Cheilion (ch)
The point located at each labial commissure
Superaurale (sa)
The highest point on the free margin of the auricle
Subaurale (sba)
The lowest point on the free margin of the ear lobe
Tragion (t)
The notch on the upper margin of the tragus
Methods
The measurements used were selected to determine the morphologic characteristics of the craniofacial complex and were conducted
according to the standard procedure described by Farkas.8 Thirty-one
direct measurements (25 single and 3 paired) were performed, and a
facial index was calculated for each subject. Standard anthropometric
instruments9 were used for these measurements. Surface landmarks
(Table 1)9 were marked on the face of each person before the
measurements were taken. Each measurement was performed twice
by the same investigator and the calculated mean value was used.
The measurements that were performed are shown in Figures 1
and 2 and may be described as follows: regarding
e72
the headthe horizontal measurement made was the head

circumference (g-op-g). The vertical measurements made were
the foreheadheight (tr-g) and the extended forehead height (tr-n).
the facethe horizontal measurementsmadewerethe maxillary
surface arc (t-sn-t), the mandibular surface arc (t-gn-t), the face
width (zy-zy), and the mandible width (go-go). The vertical
measurements made were the lower face height (sn-gn), the
physiognomical face height (tr-gn), the morphologic face height
(n-gn), the physiognomical height of the upper face (n-sto), the
height of the lower third of the face (sto-gn), the special face
height (en-gn), and the special upper face height (g-sn).
the orbitsthe horizontal measurements made were the
biocular diameter (ex-ex), the intercanthal distance (en-en),
and palpebral fissure length (ex-en).
FIGURE 1. Horizontal measurements: A, face; B, orbits; C, nose; and D, labiooral region.
frequency distribution of counts was created.4 These 3 observed

frequencies for each measurement were compared with the expected
frequencies obtained from the Greek male distribution using Chisquared test for goodness of fit. The same procedure was repeated
between Greek and North American Caucasian males as well as
Greek females and their North American Caucasian counterparts.
P values 0.009 were considered statistically significant. More
precisely, values between 0.009 and 0.001 were denominated as
very significant and those <0.001 were denominated as extremely
significant. Values between 0.01 and 0.05 were not considered
significant in the current study as they are considered to represent
differences too slight to be visually distinguishable.4
To compare the face types among Greek males and females, a
Chi-squared test was used. The null hypothesis was that the distribution of the various face types would be the same in both sexes.
FIGURE 2. Vertical measurements: A, head; B, face; C, nose; D, labio-oral region
1; E, labio-oral region 2; and F, ear.
the nosethe horizontal measurement made was the nose

width (al-al). The vertical measurement was the nose height
(n-sn) and the nasal bridge inclination (nose inclination, ni).
the labio-oral regionthe horizontal measurement made was
the mouth width (ch-ch). The vertical measuments were the
medial vermilion height of the upper lip (ls-sto), the medial
vermilion height of the lower lip (sto-li), the medial height of the
cutaneous upper lip (sn-ls), the medial vertical upper lip length
(sn-sto), and the subalare-crista philtri distance (sbal-cph).
the earthe ear length (sa-sba) and the inclination of the
medial longitudinal axis of the auricle (ear inclination, ei).
In addition, for each volunteer the facial index was calculated

using the following formula: facial index n-gn/zy-zy 100. Subsequently, the faces were classified from extreme short and wide
(hypereuriprosop) to extreme long and narrow (hyperleptoprosop), as
shown in Table 2, according to the criteria defined by Martin and
Saller10 and Garson.11
For comparison of the anthropometric facial features of the
Greek cohort with those of North American Caucasians, data from a
previous study of Farkas et al4 were used. In the latter study,
14 measurements had been performed. These were compared with
the corresponding measurements in the current study.
Statistical Methods
Initially, to compare Greek males with Greek females, the distribution of the Greek males for a given measurement was dissociated
into 3 categories: one central category spanning 2 standard deviation (SD) to 2 SD around the mean value. The other 2 categories
were entitled, one as significantly larger (with values more than 2
SD from the mean) and the other as significantly smaller (with values
more than 2 SD from the mean). Thereafter, each Greek female
subjects measurement was entered in the appropriate category, and a
TABLE 2. Classification of Facial Types According to Face Index n-gn/zyzy 100.10,11
Hypereuriprosop
Euryprosop
Mesoprosop
Leptoprosop
Hyperleptoprosop
Face Types
Male
Female
x-78.9
79.083.9
84.087.9
88.092.9
93.0-x
x-76.9
77.080.9
81.084.9
85.089.9
90.0-x
RESULTS
Anthropometric Measurements of Greek Males
and Females
A complete set of anthropometric measurements of the face was
created for each individual male and female participants. The
average values and variations were calculated (Supplemental
Digital Content 1, Table E1, http://links.lww.com/SCS/A174).
Comparison Between Greek Males and Females

When comparing the mean values of those measurements
between Greek males and females (Supplemental Digital Content
2, Table E2, http://links.lww.com/SCS/A175), some significant
differences were observed. The forehead height (tr-g) was extremely significantly smaller in females (P < 0.0001). Of the 7 vertical
measurements of the facial region, 6 showed similar values among
Greek males and females, whereas only the morphologic face height
(n_gn) was significantly smaller (P 0.0024) in females. Of the
4 horizontal measurements of the facial region, the mandibular
width (go-go), the maxillary surface arc (t-sn-t), and the mandibular
surface arc (t-gn-t) were all significantly smaller (P 0.0079,
P 0.0017, and P < 0.0001, respectively) in females.
Although in Greek males the eyes were slightly wider and spaced
further apart, and the nose longer, broader, and with greater inclination,
the differences were not statistically significant between the sexes.
The mouth was slightly wider in Greek males and situated a little
inferiorly in respect to Greek females. The upper lips were slightly
thicker in Greek females, whereas the lower lips were thicker in
Greek males. Statistically significant differences, however, were
noted neither in the vertical measurements nor in the horizontal
measurements of the labio-oral region between the sexes.
The ears were slightly longer and with greater inclination in
males, but statistically these differences were also nonsignificant.
Comparison Between Greek and North

American Caucasians
The mean values of the 14 anthropometric measurements of the
Farkas et al study4 in the Greek male and female cohorts were
compared with those of their North American Caucasian counterparts
(Supplemental Digital Contents 3 and 4, Tables E3 and E4, http://
links.lww.com/SCS/A176 and http://links.lww.com/SCS/A177).
Only horizontal facial measurements appeared to be significantly
different. Both horizontal facial measurements, the face width (zy-zy)
and mandible width (go-go), were definitely much greater (both
P < 0.0001) in Greek males. The only significant difference between
the female populations was that the mandible width (go-go) was
significantly greater (P < 0.0001) in Greek females.
e73
FIGURE 3. Face types in young adult Greek males and females.
The differences in forehead height, in the 3 vertical measurements of the facial region, in the nasal measurements, in mouth
width, and in ear length were not statistically significant.
Face Types
The most common face types were the hyperleptoprosop and
leptoprosop (long and narrow) for men and the mesoprosop (medium) for women (Fig. 3-Graph 1). Differences between the distribution of the various face types among sexes were statistically
significant (P 0.0266). In general, hypereuryprosop and euryprosop (short and wide) were found to be the least common face type in
the Greek population.
DISCUSSION
In disciplines that involve facial aesthetics it is important that the
clinicians understand the unique and detailed morphology of their
patients ethnic group before suggesting any treatment.12 More and
more patients are seeking to maintain their specific ethnic facial
features during reconstructive and aesthetic procedures. The availability of facial norms is of critical importance. Without these norms,
there is a risk of misdiagnosing, incorrect treatment planning, and
unfavorable surgical outcome in patients of different ethnic origins.
Moreover, these facial norms can be used as a guide to understand the
differences in facial profile among different ethnic groups.
It is an undisputed fact that the face differs among different
ethnicities. Farkas et al1 concluded that African-American and
North American Caucasian craniofacial complexes possess extensive and substantial differences. The most frequently observed
differences were observed in the nasal and orbital regions in both
sexes. In a study by Choe et al,2 the anthropometric measurements
of Korean-American women were very different from those of
North American Caucasian women. In another study, it was found
that female Indian-Americans differ from North American Caucasian females in 25 out of the 30 facial measurements that were
studied.3 These data support the need for further research to obtain
ethnic group-specific facial measurements.
Differences between ethnic groups within the Caucasian population have been demonstrated in several studies.13 Because facial
norms seem to be specific for each ethnicity, using the North
American Caucasian population data for all Caucasians will most
probably result in unsatisfactory outcome in aesthetic and reconstructive surgery. Unfortunately, sufficient facial anthropometric
data are not available for all ethnic groups, one of which is the Greek
population. In the current study, numerous anthropometric measurements were selected to give optimal information of the craniofacial
complex in modern Greek adults (Supplemental Digital Content 1,
Table with anthropometric measurements of Greek young adult
population). The observation of highly significant anthropometric
differences, that is differences in facial and mandibular width,
between Greek and North American Caucasians (Supplemental
Digital Contents 3 and 4, Tables with comparisons of anthropometrics of Greek males and females with North American Caucasian
e74
males and females) support the need of our study to compile a detailed
anthropometric profile of the face of Greek males and females.
The facial norms do not only differ among ethnic groups, but
also between sexes. As in accordance with other studies,1 4 in our
cohort the vast majority of anthropometric facial measurements
were greater in males, except for the medial vermilion height of the
upper lip which was greater in females (Supplemental Digital
Content 2, Table with comparison of anthropometrics of Greek
females with Greek males). Nevertheless, in the current study, most
differences were statistically nonsignificant. The statistically significant differences between the sexes were limited to the head and
face, whereas the orbits, the nose, the ears, and the labio-oral region
did not express major differences. Greek males had a significantly
wider lower face (mandibular width) with a significant greater
maxillary and mandibular surface arc, as well as a significantly
higher forehead and morphologic face. Because face index
expresses the relationship between the height of the face and its
width, Greek males consequently exhibited, in general, a higher
facial index than their female counterparts.
Besides assessment of the mean values of these anthropometric
parameters of the face and the differences between sexes and races,
it might be of importance to assess whether significant interindividual variations in these groups exist. In our Greek cohort, the
standard deviation was <10% of the mean value in the vast majority
of the 33 anthropometric measurements for both males and females
(Supplemental Digital Content 1, Table with anthropometric
measurements of Greek young adult population). This means that
for most facial features there were no major differences among
Greek individuals of both sexes. For both males and females, the
variations in forehead height (tr-g: standard deviation being 15%
and 12% of the mean value, respectively; tr-n: 13% and 11%), in
nasal bridge inclination (ni: 16% and 20%), in all vertical measurements in the labio-oral region (11%19% and 12%17%), and,
especially, in ear inclination (ei: 29% and 29%) were more pronounced. The existence of such variations in the latter parameters
may also be taken into account in the planning of aesthetic and
reconstructive surgery of the face in Greeks.
In this study, we demonstrated that with simple anthropometric
measurements it is possible to objectively create a detailed standard
facial profile of Greek males and females. As was to be expected, this
facial profile is significantly different between males and females.
For each sex, the divergences from the standard profile, however,
were in general relatively small, whereas variations in nasal bridge
and ear inclination were more pronounced. We also objectively
demonstrated that some significant facial differences exist between
sexes as well as between Greek and North American Caucasians.
The differences in normative facial anthropometric data
represent the specific facial features of ethnic groups and sexes.
Databases of such measurements should be created for all different
ethnic male and female populations to assess the differences in
facial morphology among those groups. The existence of more
pronounced interindividual variation in certain measurements
should be noted. Consideration of these specific facial features
will support the plastic surgeon in his decision as to the degree to
which certain features may be created or altered in either reconstructive or aesthetic surgery, thus assuring an optimal outcome.14
In such a way, the anthropometric data provided in the current study
offer a guide in quantitative analysis of the face of Greeks undergoing plastic and reconstructive surgery.
CONCLUSIONS
This study supports that with multiple simple anthropometric
measurements in a cohort, it is possible to objectively create a
normative facial profile of a cohort. In this study, a database of
#
detailed anthropometric measurements of the face of Greek males

and females was established. The need for such a study is supported
by the observation of some statistically significant differences when
measurements were compared with those of North American
Caucasians. Moreover, visually striking differences between the
sexes were noted. This database could be used by surgeons for the
benefit of the patients that are scheduled for reconstructive and
aesthetic facial operations to provide them with an aesthetically
harmonic appearance.
ACKNOWLEDGMENTS
The authors thank and dedicate this work to Prof Dr Vasilios Zacharopoulos, ex-vice president of the Technological Educational Institute
of Crete. Without his support and contribution, this essay would have
been impossible to be complete. Furthermore, the authors thank Mrs
Eugenia Bolbasis for the linguistic review of this manuscript.
oncocytic schneiderian papilloma. Surgical removal was recommended to the patient; however, he refused this option. Therefore,
a radiation of 64 gray (Gy) was administered in 32 daily doses (2 Gy
daily) over 45 days using intensity-modulated radiotherapy. Four
months after radiotherapy, the tumor had disappeared. Two years
postradiotherapy, the patient was healthy, without tumor recurrence
or the development of orbital complications. The authors suggest
that radiotherapy could be an adjuvant or definite treatment
modality for patients of oncocytic schneiderian papilloma unsuitable for complete surgical removal, or those associated with a high
risk of surgery-related complications.
Key Words: Nasal cavity, orbit, papilloma, radiotherapy,

schneiderian
REFERENCES
1. Farkas LG, Katic MJ, Forrest CR. Comparison of craniofacial
measurements of young adult African-American and North American
white males and females. Ann Plast Surg 2007;59:692698
2. Choe KS, Sclafani AP, Litner JA, et al. The Korean American womans
face: anthropometric measurements and quantitative analysis of facial
aesthetics. Arch Facial Plast Surg 2004;6:244252
3. Husein OF, Sepehr A, Garg R, et al. Anthropometric and aesthetic
analysis of the Indian American womans face. J Plast Reconstr Aesthet
Surg 2010;63:18251831
4. Farkas LG, Katic MJ, Forrest CR, et al. International anthropometric
study of facial morphology in various ethnic groups/races. J Craniofac
Surg 2005;16:615646
5. Vegter F, Hage JJ. Clinical anthropometry and canons of the face
in historical perspective. Plast Reconstr Surg 2000;106:10901096
6. Zacharopoulos GV, Manios A, de Bree E, et al. Neoclassical facial
canons in young adults. J Craniofac Surg 2012;23:16931698
7. Song WC, Kim JI, Kim SH, et al. Female-to-male proportions of the
head and face in Koreans. J Craniofac Surg 2009;20:356361
8. Farkas L. Anthropometry of the Head and Face. 2nd ed. New York, NY:
Raven Press; 1994
9. Farkas LG. Anthropometry of the Head and Face in Medicine.
Amsterdam: Elsevier; 1981
10. Martin R, Saller K. Lehrbuch der Anthropologie. Stuttgart: Fischer;
1957
11. Garson JG. The Frankfurt Craniometric Agreement, with Critical
Remarks Thereon. London: Hakeison and Sons; 1884
12. Turner WN. Three Dimensional Comparison of Facial Morphology of a
Caucasian American Population and a Native Brazilian Population.
Birmingham, AL: University of Alabama at Birmingham; 2012
13. Farkas LG. Accuracy of anthropometric measurements: past, present,
and future. Cleft Palate Craniofac J 1996;33:1022
14. Fang F, Clapham PJ, Chung KC. A systematic review of interethnic
variability in facial dimensions. Plast Reconstr Surg 2011;127:874881
Oncocytic Schneiderian
Papilloma of the Sinonasal Tract
Treated With Radiotherapy
Ju Wan Kang, MD, Young Suk Kim, MD,y
Jeong Hong Kim, MD, and Gwi Eon Kim, MDy
Abstract: A 66-year-old man visited the ophthalmology department due to epiphora and was subsequently diagnosed with
#
chneiderian papilloma (SP) is a benign tumor, which develops

in the schneiderian epithelium of the sinonasal tract.1 SP is
categorized into 3 subtypes: exophytic, inverted, and oncocytic.2 It
accounts for 0.5% to 4% of sinonasal tumors1; exophytic and
inverted types are more common than oncocytic forms. Oncocytic
schneiderian papilloma (OSP) is the rarest type of SP comprising
3% to 5% of all patients.3,4 Due to the rarity of OSP, its clinical
features and treatment strategies are poorly defined compared
with inverted papilloma (IP). A previous study demonstrated a
similar rate of recurrence and malignant changes between OSP
and IP.4 However, Yang et al reported a higher incidence of
malignant changes associated with OSP (10% 17%) compared
with IP (5%10%).5
Endoscopic surgical extirpation has been the treatment of choice
for OSP.3,4 A previous study reported a local OSP recurrence of
33% to 40%.6 However, Karligkiotis et al documented a mere 6%
recurrence (2 out of 33 patients) through an endoscopic approach.3
OSP does not usually affect adjacent structures; however, a patient
with OSP involving orbital and intracranial structures has been
reported.4 In this case, the patient was treated by endoscopic tumor
removal.4 There is a lack of consensus regarding the role of
radiotherapy in OSP treatment. While the role of radiotherapy in
patient with IP is also not clear, it has been utilized in patients with
inoperable tumors or residual tumors following surgical treatment.
In addition, another report has shown that radiotherapy could
represent a suitable treatment option for IP.7 However, there have
been no reports on the role of radiotherapy in the treatment of OSP.
Here, we report our recent experience of a patient with OSP treated
with radiotherapy.
From the Department of Otorhinolaryngology; and yDepartment of
Radiation Oncology, Jeju National University Hospital, Jeju, Korea.
Address correspondence and reprint requests to Jeong Hong Kim, MD,
Department of Otorhinolaryngology, Jeju National University School of
Medicine, 102 Jejudaehakno, Jeju 690-756, South Korea;
E-mail: jeonghongkimmd@gmail.com
Co-corresponding author: Gwi Eon Kim, MD, Department of Radiation
Oncology, Jeju National University Hospital, Aran 13gil 15, Jeju
690-767, South Korea; E-mail: gekim@jejunuh.co.kr
ISSN: 1049-2275
DOI: 10.1097/SCS.0000000000002233
e75
CLINICAL REPORT
A 66-year-old man presented at our ophthalmology department with
a headache in the left temporal area, epiphora, and orbital pain in the
left eye. He did not show any signs of visual disturbance, diplopia,
or limited orbital movement. He had undergone endoscopic sinus
surgery of the left nose for a polypoid mass 8 months previously.
However, there was no pathological report of this previous surgery,
and he did not have a history of any other disease. Contrastenhanced computed tomography (CT) revealed a well-enhanced
soft tissue mass in the left ethmoid sinus and nasal cavity, extending
into the extraconal space of the left orbit and the left nasolacrimal
duct (Fig. 1A). The noncontrast CT images from the patients
previous surgery were reviewed; this revealed a huge mass filling
the ethmoid sinus and nasal cavity. At that time, destruction of
lamina papyracea was suspected; however, there was no intraorbital
extension observed. A punch biopsy was performed under local
anesthesia. Histologic examination revealed an exophytic papillary
growing tumor with a long delicate fibrovascular core. The epithelium is multilayered, 2 to 10-cell thick, composed of tall
columnar oncocytic cells. The epithelium characteristically contains many small cysts filled with mucins or neutrophils, known as
microabscesses (Fig. 1B). The findings of the present case indicated
an OSP and endoscopic surgical extirpation was therefore recommended; however, the patient refused the revision surgery. Definite
radiotherapy without surgical debulking was planned as an alternative treatment. The patient was irradiated using intensity-modulated
radiotherapy; a tumor dose of 64 gray (Gy) was administered in
32 daily fractions of 2 Gy over 45 days. During radiotherapy, the
patient did not show any treatment-related orbital complications
such as disturbed visual acuity or limited eye movement. After
irradiation of 50 Gy, CT revealed that there had been no significant
FIGURE 1. A, Contrast postoperative computed tomography (CT) image showing

a well-enhanced softtissuemassin the left ethmoid sinus and nasalcavity, extending
into the extraconal space of the left orbit and left nasolacrimal duct. B, Tumor
showing exophytic branching papillary growth with a long delicate fibrovascular
core. The epithelium is multilayered and composed of tall columnar oncocytic cells.
The epithelium characteristically contains many small cysts filled with mucins or
neutrophils known as microabscesses, indicated by the arrows (H&E, 100). C, CT
image obtained after irradiation with 50 Gy, revealing an enhanced mass similar to
that observed in pretreatment CT. D, Magnetic resonance images taken 4 mo after
radiation treatment revealing only a mucosal lesion with low signal intensity on T2weighted images; there is an absence of a definite mass lesion.
e76
change in tumor size compared with pretreatment CT (Fig. 1C).

However, magnetic resonance imaging performed 4 months after
the completion of radiotherapy failed to locate a definite mass
lesion; instead, only a mucosal lesion with low signal intensity on
T2-weighted images (Fig. 1D) was observed. These findings
suggested postradiation changes without tumor recurrence. Two
years postradiotherapy, the patient was healthy, without tumor
recurrence or the development of orbital complications.
DISCUSSION
OSP is a rare benign tumor occurring in the sinonasal tract;
endoscopic surgical management is generally considered the treatment of choice. However, invasive features and malignant changes,
such as those associated with IP, have been observed in patients of
OSP. In our patient, OSP recurred 8 months after endoscopic
removal and the tumor showed a more aggressive involvement
with orbital content. Careful endoscopic extirpation of the tumor
was recommended to the patient; however, he refused this revision
surgery. There have been no previous studies on the role of radiotherapy in OSP management, although a few studies have reported
the management of IP with radiotherapy.
Radiotherapy is generally utilized as an adjuvant treatment
modality following surgery for advanced IP, with or without
associated squamous cell carcinoma. It has been found to prevent
regional or distant failure in IP with carcinoma.8 However, in 1
study, radiotherapy was reported to be ineffective in controlling the
disease; it was only successful in 2 out of 14 patients.7 Furthermore,
some authors have highlighted the risk of carcinoma development
following radiotherapy; however, there is insufficient evidence to
suggest that there is an excessive risk in patients of IP.8 An effective
dose of radiation for IP is poorly defined; however, Strojan et al
suggested that a dose of 50 to 60 Gy may be required for patients
lacking a residual tumor or for those with only a small residual
tumor after surgery.7 Furthermore, irradiation doses of up to 70 Gy
have been required in patients without surgical debulking.7 In the
present case, 64 Gy of radiation was administered to the patient
because he refused the recommended debulking of the primary tumor.
The sufficient duration for a response to radiotherapy is also
poorly defined. Strojan et al reported the results of radiotherapy in
patients of IP. CT did not reveal a reduction in tumor size 3 months
postradiotherapy; however, the size of the tumor had decreased
6 months after radiotherapy. Continued shrinkage of the tumor, as
observed from imaging studies, was reported 1 and 2 years postradiotherapy.7 In the present patient, magnetic resonance imaging
performed 4 months after radiotherapy revealed that the size of the
tumor had decreased and only a radiation-induced mucosal change
was observed. The patient was healthy without tumor recurrence
1 year following radiotherapy. However, a longer follow-up
duration is required in patients of OSP. Karligkiotis et al reported
a recurrence of OSP 46 months postsurgery and therefore proposed
an adequate follow-up period of a minimum of 5 years.3 We could
only follow the patient for 2 years postradiotherapy because after
that, he did not return to the hospital for the regular scheduled
follow-up visits. Our patient responded well to radiotherapy within
4 months of treatment; however, the relatively short follow-up
duration is a major limitation of this study.
OSP is a benign tumor of the sinonasal tract, and the result of
endoscopic surgical treatment is generally acceptable. However, the
invasiveness and malignant changes of OSP should be considered and
the aggressive OSP or unsuitable patients for surgical treatment need
an adjuvant or alternative treatment modality. To the best of our
knowledge, this is the first patient with OSP to be successfully treated
with radiotherapy. Therefore, we suggest that radiotherapy could be a
potential adjuvant or definite treatment modality for OSP.
#
REFERENCES
1. Vorasubin N, Vira D, Suh JD, et al. Schneiderian papillomas: comparative
review of exophytic, oncocytic, and inverted types. Am J Rhinol Allergy
2013;27:287292
2. Shanmugaratnam K, Sobin LH. The World Health Organization
histological classification of tumours of the upper respiratory tract and
ear. A commentary on the second edition. Cancer 1993;71:26892697
3. Karligkiotis A, Bignami M, Terranova P, et al. Oncocytic Schneiderian
papillomas: clinical behavior and outcomes of the endoscopic endonasal
approach in 33 cases. Head Neck 2014;36:624630
4. Bignami M, Pistochini A, Meloni F, et al. A rare case of oncocytic
Schneiderian papilloma with intradural and intraorbital extension with
notes of operative techniques. Rhinology 2009;47:316319
5. Yang YJ, Abraham JL. Undifferentiated carcinoma arising in oncocytic
Schneiderian (cylindrical cell) papilloma. J Oral Maxillofac Surg
1997;55:289294
6. Kaufman MR, Brandwein MS, Lawson W. Sinonasal papillomas:
clinicopathologic review of 40 patients with inverted and oncocytic
schneiderian papillomas. Laryngoscope 2002;112 (8 pt 1):13721377
7. Strojan P, Jereb S, Borsos I, et al. Radiotherapy for inverted papilloma: a
case report and review of the literature. Radiol Oncol 2013;47:7176
8. Hug EB, Wang CC, Montgomery WW, et al. Management of inverted
papilloma of the nasal cavity and paranasal sinuses: importance of
radiation therapy. Int J Radiat Oncol Biol Phys 1993;26:6772
They were removed by a mini goblet forcep completely and

efficiently. A whole bone lid was replaced with a biological
membrane to help repair bone defect after removing procedure.
Results: The operation is about 20 minutes with endoscope and
piezoelectric device helped to save a lot of time and provided
excellent visual surgical field. Main postoperative adverse effects
were swelling, numbness, and temporal no-vitality for the first
premolar (24). Three months later, computed tomography shows the
Schneiderian membrane thinned to around 0.8 mm. The bone lid is
on its position and starts to perform synostosis. The 24 tooth is still
dysesthetic and needs time to recover.
Conclusions: Endoscopic surgery combined with a piezoelectric
device has obvious advantage of minimizing surgical injury and
providing excellent visibility of surgical field when removing longterm foreign bodies in maxillary sinus. It is efficient and protects the
residual alveolar bone.
Key Words: Endoscopic-assisted surgery, maxillary sinus,
minimally invasive, piezoelectric device, root fragment
Purpose: This report presented a patient with 2 long-term broken

roots displaced in left maxillary sinus. The residual root fragments
made the patient uncomfortable in both mind and body and interfered with prosthodontics work. The application of endoscope
combined with piezoelectric device both helps in removing the
broken roots successfully with minimally surgical injury and preserves the residual alveolar bone.
Methods: Computed tomography scans and 3-dimensional reconstructions located the broken roots. A 1.0 cm 1.5 cm rectangle
bone window on anterolateral sinus wall was opened by a piezoelectric device to place the endoscope and forcep into sinus. Two
broken roots could be observed clearly via a endoscopic screen.
t is very common for root fracturing when extracting the upper

molar with multiple and separated roots. For some anatomy
factors, broken roots could displace into the maxillary sinus accidentally if dentists elevate the roots with excessive force. Thus, it
leads to some serious complications, such as chronic sinusitis, oroantral fistula, and pain. It is more challenging to remove the broken
roots in the sinus after the extraction socket has completely healed.
There are solutions to remove the broken roots before the extraction
socket healed up, like irrigation and suction from the enlarged
extraction socket. But these methods cannot remove the broken
roots after the extraction socket had already healed up.
For long-term foreign bodies in maxillary sinus like displaced
broken roots in sinus, bone window approach is a normal way to
remove them. There are 3 main methods to open a bone window to
approach the foreign bodies in maxillary sinus: via nasal meatus; via
canine fossa; via lateral wall of maxillary sinus. To avoid big injury
caused by opening a direct vision bone window, many dentists use
endoscope to reduce the surgical injury.
In this report, 2 long-term broken roots that were located by
preoperative computed tomography (CT) were on the bottom of left
maxillary sinus. They were adhered to bone and surrounded by
incrassated Schneiderian membrane. The extraction socket had
already healed up. In order to preserve the alveolar bone, bone window
is the only way to approach those root fragments. And a special forcep
was used to separate Schneiderian membrane and remove them.
From the Department of Oral and Maxillofacial Surgery, Shanghai Ninth

Peoples Hospital, Shanghai Jiao Tong University School of Medicine,
Shanghai Key Laboratory of Stomatology, Shanghai, China.
Address correspondence and reprint requests to Chi Yang, DDS, PhD, and
Ling Yan Zheng, DDS, PhD, Department of Oral and Maxillofacial
Surgery, Shanghai Ninth Peoples Hospital, Shanghai Jiao Tong
University School of Medicine, No. 500 Qu-Xi road, 200011 Shanghai,
China; E-mail: yangchi63@hotmail.com; zhenglingyan73@163.com
This project was supported by the National Natural Scientific Foundation of
China (81100766), the Key Subject Construction Project of Shanghai
(S30206), and the Key Project of Shanghai Municipal Health Bureau
(2014035).
ISSN: 1049-2275
DOI: 10.1097/SCS.0000000000002235
A 30-year-old woman, without any systemic disease, came to the

Department of Oral Surgery, presented with frequently dull pain on
the left upper face. Her left maxillary second molar and third molar
had been extracted out 1 year ago by the reason of useless residual
crown, but some of the roots had been broken up and the root
fragments had been left inside the socket. Since then she has begun
to feel uncomfortable, and had to use antibiotics to relief the
symptoms. A CT scan helped make a 3-dimensional reconstruction
(Fig. 1) to see the locations of those root fragments. There were
2 root fragments of second molar which were around 5 mm, were at
the bottom of left maxillary sinus adhered to bone and incrassated
Schneiderian membrane, and 1 root of third molar is inside the
alveolar crest, the bottom of left maxillary Schneiderian membrane
was incrassated, about 2 to 3 mm (Fig. 1). The patient feels
uncomfortable both physically and mentally; meanwhile, she also
Removal of Long-Term Broken

Roots Displaced Into the Maxillary
Sinus by Endoscopic Assistant
Qi Man Gao, MD, Chi Yang, DDS, PhD,
Ling Yan Zheng, DDS, PhD, and Ying Kai Hu, MD
CLINICAL REPORT
e77
FIGURE 1. A and B, The preoperation computed tomography film showed

that 2 roots of second molar were at the bottom of left maxillary sinus, and 1 root of
third molar was inside the alveolar bone. C and D, A 3-dimensional reconstruction.
FIGURE 3. A, The Schneiderian membrane was cut for 3 mm and

blunt separated. B, The 2 broken roots seated in the bottom of left sinus,
surrounded by thickened Schneiderian membrane. C and D, Using the mini
goblet forcep to separate the membrane and remove one of the broken roots.
needs implant prosthesis. After eliminating some contraindications,

a surgery has been arranged for her.
After the disinfection procedures and local anesthesia, a long
mucosa incision was made from the left upper lateral incisor area to
the first molar area by an electrosurgical knife. A piezoelectric device
was used to open a 1.0 cm 1.5 cm rectangular bone window on
the anterolateral maxillary sinus wall above the premolar area; the
Schneiderian membrane was exposed after peeling off the bone lid,
which was soaked in the sterile normal saline (Fig. 2). The Schneiderian membrane was cut for 3 mm and blunt separated, thus revealing the 2 broken roots that were shown clearly by the endoscope
seated in the bottom of left sinus. The 2 broken roots were adhered to
bone and surrounded by incrassated Schneiderian membrane.
Although the Schneiderian membrane around the broken roots
was incrassated, it is still healthy without hyperemia. The mini goblet
forcep has been used to separate the root fragments from the
membrane, and the fragments were extracted with little resistance
(Fig. 3). During the surgery, plenty of irrigation of the sinus with cold
(48C58C) sterile normal saline was performed to keep visual field
clear and clean up any remaining infective materials. To close the
bone window and minimize the injury, a bio-gide collagen membrane
was secured with submucosa tissue first. Then, the bone lid was
replaced back to restore the integrity of anterolateral wall of sinus.

Finally, the incision was sutured (Fig. 4). Local pressure dressing
under left zygoma has been applied to reduce postoperation swelling.
Two weeks later, patient came back for follow-up. She bore
swelling and pain on left face for 4 days, numbness for 9 days, and
gradually reduced mucus in nose for 1 week. But, the sores of digital
technology automatic pulp vitality tester on left upper tooth showed
there is no pulp vitality on the 24 tooth.
Three months later, she came again for further consultation. The
thickness of the left maxillary Schneiderian membrane is normal
(around 0.8 mm). The bone lid is on its position and starts to perform
synostosis. The vitality score of the 24 tooth turns to 64, indicating
dysesthesia on the 24 tooth. And the intraoral x-ray film showed
nothing wrong with the 24 tooth (Fig. 5).
There are 2 major methods classified by approach ways, to remove

the foreign bodied displaced into maxillary sinus: crestal approach,
bone window approach via adjacent structure. Suction through
crestal1 is used to remove the broken roots before the extraction
healed up. It is convenient and low cost. But the alveolar bone will
FIGURE 2. A, Intraoral condition before operation. B, A long incision was made.

C, A 1.0 cm 1.5 cm rectangular bone window on the anterolateral maxillary
wall. D, The Schneiderian membrane was exposed after peeling off the bone lid.
FIGURE 4. A and B, Removal of another root. C, A bio-gide collagen membrane

was secured with submucosa tissue to cover the window, and the bone lid was
replaced; D, Suture up.
e78
DISCUSSION
FIGURE 5. A, The 2 removed broken roots; B, the interoral X-ray film took 3 mo
after operation showed nothing wrong with the left upper first premolar; C,
Computed tomography 3 mo after operation shows the thickened Schneiderian
membrane has been thinned (around 0.8 mm); D, A 3-dimensional
reconstruction 3 mo after operation, the bone lid is on its position and starts to
perform synostosis, healing with adjacent bone tissue.
be damaged by the socket-enlarging procedure, which influences

the healing procedure and residual bone height, interferes with
prosthodontics work. Therefore, the crestal approach method is not
a good solution to remove the long-term broken roots after the
extraction socket had already healed up.
Opening a bone window via an adjacent structure is an efficient
method to both remove the foreign body and preserve the alveolar
bone. The bone window could be located on the lateral maxillary
sinus wall,2 canine fossa,3 or middle/inferior nasal meatus.4 The
traditional CaldwellLuc is approach3 from canine fossa but without any bone grafting to restore the bone defect on the sinus wall.
The transnasal endoscopic approach5 can avoid bone defect on the
sinus wall, but it requires specific training and equipment. Besides
these 2 approach windows are too far from the located broken roots
that increase the difficulty in surgery.
The traditional lateral window approach normally opens a bone
window on lateral maxillary sinus by power drill without restoring
the bony defect. Although the traditional way can expose the broken
roots to the operators visual field directly, which helps remove the
roots under clear vision, the big surgical injury, long operating time,
severe reactive swelling, and high risk of nerve damage are still big
problems. Nowadays, there are some reports about piezoelectric
device6 used on the removal procedure to reduce the surgical injury.
Furthermore, the application of endoscope7 is a great breakthrough to reduce the surgical injury and risk, and to protect nearby
vital structure.8 Operators can see the inside of sinus clearly by
endoscopic via a smaller bone window. Thus, the endoscope is widely
used to reduce surgical injury.911 Albu12 reported a patient with
double-barrel approach to the removal of a long-term dental
implant from maxillary sinus, using a trocar rotated to penetrate
2 small windows on the anterior wall of maxillary sinus, and put into
endoscope and forcep through the trocars to remove the foreign body.
In this case, the 2 long-term broken roots were on the bottom of left
maxillary sinus. They were adhered to bone and surrounded by
incrassated Schneiderian membrane. And the extraction socket has
already healed up. To prepare for the implant prosthesis, approaching
way with 1 little bone window about 1.0 cm 1.5 cm at the anterolateral wall of the left sinus has been cut up. The diameter of the
endoscope is 5 mm; the diameter of the suction hose is 5 mm; and the
diameter of the mini goblet forcep is 2 mm. Therefore, the endoscope,
suction hose, and a special instrumentthe mini goblet forcep that
#
used to separate and remove the root fragments from incrassated

membrane could be placed into the sinus simultaneously.
The Schneiderian membrane has the ability to recover as long as
the bone under it is consecutive. Moreover, during the surgery
procedure, the endoscope showed that the Schneiderian membrane
around the roots was incrassated, but looks healthy with no hyperemia. Thus, the covered membrane was separated from root fragments, without any disposal like scraping. Only copious sinus
irrigation was performed.
While for the bone lid, it was cut off by a piezoelectric device,
and kept in normal saline, a biological membrane is used to close
the window, and then, replace the bone lid, suture up the mucosal
incision. The replaced bone lid will help restore the integrity of
anterolateral wall of sinus and promote the recovery procedure with
osteoinductivity. There are some other methods to immobilize the
bone lid, such as immobilizing the bone lid with adjacent bone by
titanium miniplates6 or absorbable suture material13 through previously prepared holes. But, drilling holes may increase the risk of
damage to the adjacent nerves and vessels.
During the surgery procedure, the application of endoscope and
piezoelectric device helped save a lot of time and provided excellent
visual surgical field. The piezoelectric device creates a smooth and
clean bone window rapidly with little bleeding, and preserves a
whole bone lid, which was used to restore the bone defect later.
Endoscope helps locate the root fragment and keep the vision field
clear. The whole time of operation was about 20 minutes. It is
effective and efficient with minimally invasive.
Three months later, CT shows the thickened Schneiderian membrane has been thinned, being around 0.8 mm. The bone lid is on its
position and starts to perform synostosis. But, the pulp vitality test
indicates that the 24 tooth is still dysesthetic and needs time to go back
to normal. The intraoral x-ray shows a complete tooth of 24, which
demonstrates that the cause of dysesthesia should be injury occurred
on superior alveolar nerve. The distinction of superior alveolar nerve
does have some regulation.14 But there is some variation on nerve
distinction, and we cannot prognose it on preoperative CT. It is hard to
avoid such nerve injury15 but we should make force to minimize the
surgical injury and reduce the risk.
CONCLUSIONS
An anterolateral maxillary sinus window approach surgery assisted
by endoscope combined with a piezoelectric device is an efficient
and effective method to remove long-term broken roots or other
foreign bodies in maxillary sinus. It provides excellent visibility of
surgical field, avoids big damage, and preserves the alveolar bone
for implant prosthesis later. The replaced bone lid helps restore the
integrity of anterolateral sinus wall.
REFERENCES
1. Nakamura N, Mitsuyasu T, Ohishi M. Endoscopic removal of a dental
implant displaced into the maxillary sinus: technical note. Int J Oral
2. Hu YK, Yang C, Xu GZ, et al. Retrieval of root fragment in maxillary
sinus via anterolateral wall of the sinus to preserve alveolar bone.
J Craniofac Surg 2015;26:e81e84
3. Huang IY, Chen CM, Chuang FH. Caldwell-Luc procedure for retrieval
of displaced root in the maxillary sinus. Oral Surg Oral Med Oral Pathol
Oral Radiol Endod 2011;112.6:e5963
4. Kim JW, Lee CH, Kwon TK, Kim DK. Endoscopic removal of a dental
implant through a middle meatal antrostomy. Br J Oral Maxillofac Surg
2007;45:408409
5. Kitamura A. Removal of a migrated dental implant from a maxillary sinus
by transnasal endoscopy. Br J Oral Maxillofac Surg 2007;45:410411
6. Bacci C, Sivolella S, Brunello G, et al. Maxillary sinus bone lid with
pedicled bone flap for foreign body removal: the piezoelectric device.
Br J Oral Maxillofac Surg 2014;52:987989
e79
7. Giovannetti F, Priore P, Raponi I, et al. Endoscopic sinus surgery in

sinus-oral athology. J Craniofac Surg 2014;25:991994
8. Jacob KJ, George S, Preethi S, et al. A comparative study between
endoscopic middle meatal antrostomy and Caldwell-Luc surgery in the
treatment of chronic maxillary sinusitis. Indian J Otolaryngol Head
Neck Surg 2011;63:214219
9. Felisati G, Lozza P, Chiapasco M, et al. Endoscopic removal of an
unusual foreign body in the sphenoid sinus: an oral implant. Clin Oral
Implants Res 2007;18:776780
10. Chiapasco M, Felisati G, Maccari A, et al. The management of
complications following displacement of oral implants in the
paranasal sinuses: a multicenter clinical report and proposed treatment
protocols. Int J Oral Maxillofac Surg 2009;38:12731278
11. Ramotar H, Jaberoo MC, Koo Ng NK, et al. Image-guided, endoscopic
removal of migrated titanium dental implants from maxillary sinus: two
cases. J Laryngol Otol 2010;124:433436
12. Albu S. The double-barrel approach to the removal of dental implants
from the maxillary sinus. Int J Oral Maxillofac Surg 2013;42:15291532
13. Biglioli F, Goisis M. Access to the maxillary sinus using a bone flap on a
mucosal pedicle: preliminary report. J Craniomaxillofac Surg
2002;30:255259
14. Robinson S, Wormald PJ. Patterns of innervation of the anterior maxilla:
a cadaver study with relevance to canine fossa puncture of the maxillary
sinus. Laryngoscope 2005;115:17851788
15. Robinson SR, Baird R, Le T, et al. The incidence of complications after
canine fossa puncture performed during endoscopic sinus surgery. Am J
Rhinol 2005;19:203206
Synchronous Basal Cell

Carcinoma of the Inferior EyelidCombined Surgical Approach
for Single-Stage Ablation
Yordan Petrov Yordanov, MD, PhD and Aylin Shef, MDy
Abstract: Basal cell carcinoma (BCC) is the most common
cutaneous malignancy which can result in significant patient morbidity, especially when delicate periocular skin zone is affected.
Late diagnosis of these tumors determines more aggressive surgical
approach, which often requires combination of different reconstructive techniques and strategies. The authors report on a clinical
case of a 67-year-old woman with synchronous BCC of the lower
eyelid treated surgically in one stage by combining basic principles
of both the aesthetic and reconstructive eyelid surgery. A special
emphasis is put on the 5-step operative technique the authors have
applied preserving form and function.
Key Words: Basal cell carcinoma, lower eyelid, single-stage
reconstruction, synchronous
From the Unit of Plastic Surgery and Burns, Military Medical Academy;
and yDepartment of Dermatovenereology and Allergology, Military

Medical Academy, Sofia, Bulgaria.
Address correspondence and reprint requests to Yordan Petrov Yordanov,
MD, PhD, Unit of Plastic Surgery and Burns, Military Medical Academy, 3 Georgy Sofiisky Blvd, 12th Floor, 1606 Sofia, Bulgaria;
E-mail: yordanov_vma@abv.bg
ISSN: 1049-2275
DOI: 10.1097/SCS.0000000000002236
e80
asal cell carcinoma (BCC) is the most common cutaneous

malignancy worldwide accounting for approximately 80% of
all skin cancers.1 Although mortality is rare, locally aggressive BCCs
can result in significant patient morbidity, especially when delicate
zones of the head are affected.2 Periocular skin zone, in particular, is a
common site of neoplastic lesionsapproximately 5% to 10% of all
skin cancers occur in the eyelids.3 Late diagnosis of these tumors
determines more aggressive surgical approach which often requires
combination of different techniques and strategies to restore the
normal shape of the eyelids and diminish the functional impairment.
We present a woman with synchronous BCC of the lower eyelid
treated surgically in one stage by combining basic principles of both
aesthetic and reconstructive eyelid surgery.
CLINICAL REPORT
A 67-year-old Caucasian woman referred to us for the evaluation of
a lesion on her right lower eyelid which had been present for several
years. The patient had a strong history of chronic sun exposure and no
family history of skin malignancies. Clinical examination revealed a
1.8 cm 1.2 cm exophytic pigmented tumor engaging the margin of
the right lower eyelid of its middle to medial third on approximately
40% of its total length. Lateral to the described lesion, a second one
was found. The later was a pigmented plaque with oval shape
occupying the pretarsal eyelid skin with no involvement of the margin
and eyelashes (Fig. 1A). It appeared a few months after the bigger one
and was enlarging much more slowly over time. A vision field
obstruction on a downward gaze was detected. No lymphadenopathy
of the regional lymph nodes was found. The 2 lesions were clinically
interpreted as a synchronous pigmented BCC, and a surgical treatment was considered.
Under general anesthesia, a surgical excision of the 2 lesions and
total eyelid reconstruction was carried out in 5-step intervention. In the
first step, the large lesion was removed by pentagonal edge resection
with 2 mm of margin because of the proximity to the lacrimal punctum
medially. The shape of the excision was perpendicular to the lid margin
and then converged to a point in the midline of the planed fullthickness defect to preserve vertical height of the eyelid.
In the second step, an excision of the smaller lesion was
intended. As the lesion was situated at 2 mm from the eyelashes
line, a subciliary incision in the lateral remaining part of the
resected eyelid was performed. A palpebral skin flap was elevated
distally to the orbital rim by meticulous dissection in subcutaneous
plane applying the conventional skin-flap-only aesthetic blepharoplasty technique. Then, the tumor was excised by elliptical excision from the cranial edge of the dissected skin flap.
To reduce the excessive tension, in the third step, a cantholysis
of the lateral canthal tendon was performed, and a minimal recruit
of temporal skin was also executed similarly to the Tanzel rotation
flap reconstructive technique described elsewhere (Fig. 1B).4 In the
fourth step, the full-thickness defect was meticulously repaired in
layers living the anterior lamella still open. In the final step, the
inferior-lateral cutaneous eyelid flap was lifted and sutured in
tension-free manner to the remaining part of the anterior lamella
FIGURE 1. A, Two synchronous pigmented BCC on the same eyelid. B, Surgical

defect before the reconstruction. Two holding sutures are lifting the medial and
lateral edges of the full-thickness defect remaining after the ablation of the
exophytic BCC. Note the inferior-lateral cutaneous flap and the incision
extending into the temporal zonethe lateral BCC was removed by a marginal
excision from the cranial edge of the flap. C, Final result at sixth postoperative
month. No functional impairment was detected. BCC, basal cell carcinoma.
of the medial portion of the eyelid and then to the lid margin
applying the principles of the aesthetic eyelid skin lifting.
Surgical recovery was uneventful with no complications.
At postoperative sixth month, a clinical evaluation revealed an
excellent aesthetic and functional outcome (Fig. 1C).
dorsal augmentation material was identified. It was circumferentially enclosed with bony material and hypertrophied bony lesion
induced hump on the mid portion of nasal dorsum. During operation, the authors found it was the calcified capsule of silicone
implant, and the calcification was surrounding the whole implant
material.
DISCUSSION
Basal cell carcinoma is the most frequent malignant tumor of
the eyelid, and the sun exposure is reported to be a main risk
factor.3,5 The challenge of eyelid reconstruction revolves
around the restoration of complex eyelid anatomy and function,
as well as recreating a natural aesthetic appearance. In our case, we
have successfully applied a systematic approach taking into
account the principles of both reconstructive and aesthetic
blepharoplasty techniques.6
In addition to the challenging presurgical planning, in our
patient we faced the difficulty of treating a synchronous BCC of
the lower eyelid with no technical possibilities to apply a Mohs
micrographic surgical approach which should be indicated in such
difficult cases. We had to deal with very close margins in the
extremely gentle area of the lacrimal punctum. Nevertheless,
the intraoperative frozen section shows free of tumor infiltration
section margins and the permanent sections confirmed that.
To the best of our knowledge, this is the first report of synchronous BCC of the lower eyelid reconstructed in a single-stage
intervention.
Key Words: Calcification, complication, rhinoplasty, silicone
sian nose usually require augmentation rhinoplasty rather than

reduction rhinoplasty.1 The need for augmentation material that
is readily available, not associated with donor-site morbidity, offers
ease of sculpting, and has smooth contours has led to the introduction of alloplastic implants. Among various alloplastic implant
materials, silicone rubber is usually used for augmentation of nasal
dorsum, especially in Asian countries.2 Previous studies suggested
that silicone implants are appropriate material for augmentation
rhinoplasty with long-standing results and low complication rates.
Compared with silicone implant used in breast augmentation
surgery, calcification of nasal silicone implant is rarely reported.3
Recently, we experienced a case of silicone implant which was
circumferentially enclosed with calcified capsule. Preoperatively, it
was misdiagnosed as an artificial bony substitute because the
surrounding calcification was very regular, and encircled the whole
implant material.
REFERENCES
CLINICAL REPORT
1. Ceradini DJ, Blechman KM. Dermatology for plastic surgeons II

cutaneous malignancies. In: Thorne CH, Chung KC, Gosain AK,
Gurtner GC, Mehrara BJ, Rubin JP, Spear SL, eds. Grabb and Smiths
Plastic Surgery. Philadelphia, PA: Lippincott Williams & Wilkins; 2014:
A 53-year-old woman presented with the complaint of external

nasal contour change. She said that she had rhinoplasty using the
bone of shark 30 years ago. Dorsal nasal hump was observed, and
others were not remarkable. Computed tomography (CT) scan
showed that previously inserted L-shaped augmentation material
was entirely covered with the capsule showing high signal intensity
suggesting bony material (Fig. 1A, B). Preoperatively, we could not
confirm which was that material, and speculated that might be part
of animal bone or artificial bony institute, because the patient
insisted shark bone was inserted in her nose, and the calcification
was very regularly enclosing the augmentation material. We
decided to perform revision rhinoplasty, and surgery was performed
under local anesthesia with intravenous sedation. Septal cartilage
for columellar strut and nasal tip onlay graft was harvested using
Killian approach. After bilateral alar marginal and inverted V
transcolumellar incisions, we found that the previously inserted
augmentation material was L-shaped silicone that was fully capsulated with calcified tissue (Fig. 1C, D). We completely removed the
material with calcified capsule and performed revision rhinoplasty
using type 1 silastic implant and septal cartilage. Removal of whole
capsule and previous implant material was enough for the correction of hump, and strut graft and only graft was performed for tip
115126
2. Lasso JM, Yordanov YP, Pinilla C, et al. Invasive basal cell carcinoma
in a xeroderma pigmentosum patient: facing secondary and tertiary
aggressive recurrences. J Craniofac Surg 2014;25:e336e338
3. Cook BE Jr, Bartley GB. Treatment options and future prospects for the
management of eyelid malignancies: an evidence-based update.
Ophthalmology 2001;108:20882098
4. Sassoon EM, Codner MA. Eyelid reconstruction. Oper Tech Plast
5. Deprez M, Uffer S. Clinicopathological features of eyelid skin tumors.
A retrospective study of 5504 cases and review of literature. Am J
Dermatopathol 2009;31:256262
6. Trussler AP, Rohrich RJ. MOC-PSSM CME article: blepharoplasty. Plast
Reconstr Surg 2008;121 (1 suppl):110
Circumferential Calcification of
Silicone Implant Misunderstood
as a Bony Substitute
Sae Bin Lee, MD and Hyun Jin Min, MD, PhDy
Abstract: Silicone implant is known to be safe and easy to handle,
and frequently used in Asian rhinoplasty. Compared with breast
implant, complication studies about silicone calcification used
in rhinoplasty are very limited. Recently, the authors experienced
an interesting patient who underwent revision rhinoplasty in our
institution. Based on preoperative images, previously inserted
#
From the Unique Aesthetic Clinic; and yDepartment of OtorhinolaryngologyHead and Neck Surgery, Chung-Ang University College of
Medicine, Seoul, Korea.
Address correspondence and reprint requests to Hyun Jin Min, MD, PhD,
Department of OtorhinolaryngologyHead and Neck Surgery, ChungAng University College of Medicine, 102, Heukseok-ro, Dongjak-gu,
Seoul 156-755, South Korea; E-mail: jjinient@gmail.com
ISSN: 1049-2275
DOI: 10.1097/SCS.0000000000002237
e81
FIGURE 1. Coronal (A) and sagittal (B) images of computed tomography scans.
Bony material was identified surrounding implant material, and hypertrophied
bony material was observed on the mid portion of nasal dorsum. During
operation, L-shaped silicone implant material (C) and calcified capsule that was
circumferentially encircling the silicone (D) were identified and removed.
Calcification on the surface of silicone implant itself was also observed.
plasty. The patient was satisfied with the surgical outcome, and until
6 months, there was no reported complication.
DISCUSSION
Asian surgeons generally prefer alloplastic material for augmentation rhinoplasty, whereas Western surgeons tend to be more
comfortable with autologous materials.2 Although autologous augmentation materials have been widely used, they have several
limitations. Additional operation should be performed to get calvarial or iliac crest bone with the increased risk of gait disturbance
and intracranial complications. Infection or poor scarring could be
happened in auricular and septal cartilage harvest.4 Alloplastic
implant is known to be nontoxic, chemically stable, and show
excellent biocompatibility.5 Among various alloplastic materials,
silicone rubber can be easily carved, and does not warp, making
intraoperative handling and placement easier. These reasons enable
popular use of silicone material. In area of breast implants, the
complication regarding calcification of silicone has been continuously studied. However, in area of rhinoplasty, the study regarding
calcification of silicone implant is extremely rare. Our experience is
an interesting case report that calcification capsule was misunderstood as an animal bone or artificial bony substitute because it
regularly enclosed the whole implant.
It has been studied that calcification was occurred after 5 years
8 months of insertion.5 They suggested that longer the implanted
period, the broader the calcifications observed on gross examination.5
In another study, the most important factors affecting the calcification
of silicone implant were duration of insertion, and type of silicone.6
In our patient, it has been 30 years since previous rhinoplasty, and
longer duration might be the reason of whole capsular calcification of
silicone implant. We also found that not only the capsule, but silicone
implant itself, was also calcified (Fig. 1C).
It was reported that calcification was frequently associated with
significant pain, and firmness from capsular contracture.7 In our
patient, there was no pain, but dorsal irregularity was the only
subjective symptom. And removal of silicone with calcified capsule
diminished the dorsal hump. In our patient, calcification of silicone
implant induced dorsal irregularity and misunderstood as a bony
hump preoperatively. Although the studies about calcification
accompanied in nasal silicone implant are very rare, our case
suggests that excessive calcification could be happened in nasal
silicone implant, which could affect the external nasal contour.
Furthermore, calcification capsule surrounding the whole nasal dorsal
area could be formed as a late complication of silicone implant which
was suspected to be a foreign body in our patient. In a consideration of
revision rhinoplasty, especially in patients who previously used
silicone implant, surgeons should keep in mind that the degree of
calcification could be much more excessive than their prediction. And
the removal of whole calcified capsule including implant should be
evaluated for the correction of external nasal contour.
e82
In another study, infection was one of the most dread complications of silicone implants according to the previous study,4 but
there was no infection sign in our patient. We support the suggestion
that the degree of inflammation of calcified implant capsule
depends on the individual, rather than the duration.5
Although silicone implants are widely used, and known to be
safe, calcification or calcification-induced deformities of external
nasal contour should be considered as one of the late complications,
especially when it was inserted for a long period.
REFERENCES
1. Ahn J, Honrado C, Horn C. Combined silicone and cartilage implants:
augmentation rhinoplasty in Asian patients. Arch Facial Plast Surg
2004;6:120123
2. McCurdy JA Jr. The Asian nose: augmentation rhinoplasty with
L-shaped silicone implants. Facial Plast Surg 2002;18:245252
3. Pak MW, Chan ES, van Hasselt CA. Late complications of nasal
augmentation using silicone implants. J Laryngol Otol 1998;112:10741077
4. Erlich MA, Parhiscar A. Nasal dorsal augmentation with silicone
implants. Facial Plast Surg 2003;19:325330
5. Jung DH, Kim BR, Choi JY, et al. Gross and pathologic analysis of longterm silicone implants inserted into the human body for augmentation
rhinoplasty: 221 revision cases. Plast Reconstr Surg 2007;120:19972003
6. Peters W, Pritzker K, Smith D, et al. Capsular calcification associated
with silicone breast implants: incidence, determinants, and
characterization. Ann Plast Surg 1998;41:348360
7. Luke JL, Kalasinsky VF, Turnicky RP, et al. Pathological and biophysical
findings associated with silicone breast implants: a study of capsular
tissues from 86 cases. Plast Reconstr Surg 1997;100:15581565
A Comparison of Free Tissue

Transfers to the Head and Neck
Performed by Sugeons and
Otolaryngologists
Anthony M. Kordahi, BA, Ian C. Hoppe, MD,
and Edward S. Lee, MD
Background: The reconstruction of defects resulting from the
extirpation of head and neck neoplasms is performed by both
otolaryngology and plastic surgery services, mostly dependent on
the institution. The American College of Surgeons National Surgical Quality Improvement Program (NSQIP) provides a unique
From the Division of Plastic Surgery, Department of Surgery, Rutgers New
Jersey Medical School, Newark, NJ.
Received January 27, 2015.
Address correspondence and reprint request to Ian C. Hoppe, MD, Division
of Plastic Surgery, Department of Surgery, Rutgers New Jersey Medical
School, 140 Bergen Street, E1620 Newark, NJ 07103;
E-mail: ianhoppe@gmail.com
This article was presented at the American Society for Reconstructive
Microsurgerys Annual Meeting in Kauai, HI, January 814, 2014.
This article was presented in poster format at the 59th Annual Meeting of the
Plastic Surgery Research Council in New York, NY, March 79, 2014.
ISSN: 1049-2275
DOI: 10.1097/SCS.0000000000002240
#
opportunity to examine a predefined set of variables with regard

to free vascularized tissue transfers performed by each service.
Methods: Following institutional review board approval, the
NSQIP Participant Use Files for 2005 to 2011 were examined
for all Current Procedural Terminology codes regarding free tissue
transfer and with primary ICD-9 codes indicating a head and neck
neoplasm. Each record was examined to determine which service
performed the free tissue reconstruction and subsequent outcomes.
Results: During this time period a total of 534 flaps were performed, 213 by plastic surgery and 321 by otolaryngology. Total
hospital length of stay was 12.9 and 11.2 days for plastic surgery
and otolaryngology, respectively (P < 0.05). There were no significant differences noted between surgical site infections, wound
dehiscence, and flap failure. Patients undergoing flaps performed by
plastic surgery were significantly more likely to be on a ventilator
48 hours postoperatively (P < 0.005). Plastic surgery performed a
significantly increased number of osseous flaps compared with
otolaryngology (P < 0.05).
Conclusions: This study shows similar results with regard to free
vascularized tissue transfers when performed by plastic surgery and
otolaryngology. Slightly longer hospital stays and longer time spent
on the ventilator may be associated with the increased number of
osseous flaps performed by plastic surgery.
Key Words: Free tissue flaps, otolaryngology, plastic surgery,
comparison
eformities of the head and neck are most commonly caused by

neoplasms, trauma, and congenital defects.1 The prominent
location of these deformities and the associated severe social stigma
spurred the nascence of cosmetic and reconstructive surgery as early
as 800 BC.2 The current goal of reconstruction of the head and neck
through the use of free tissue transfer is to provide functional,
aesthetic, and psychologic enhancement to patients quality of life.
The progress of these procedures has been exponential over the past
century, carefully honed based on advancements made in microsurgical techniques, most notably the introduction of the microscope
into the operating room by Nylen and Holmgren in the 1920s.3
In 1960, Jacobson and Suarez were able to exploit the surgical
microscope in the anastomosis of small blood vessels. Throughout
the 1960s, surgeons such as Krizek and Bunke performed experiments transplanting tissues in animals, laying the groundwork for
the future use of microvascular free tissue transfers in reconstructive surgery.3 Today, free tissue transfers are considered an attractive choice for head and neck reconstruction for myriad reasons.
Microvascular free flaps allow the surgeon a wide selection of
donor sites to match the defects size and tissue needs.4 Although
overall success rates from microvascular free flaps range from 94%
to 96%, there are many risk factors that predispose patients to
postoperative complications, including but not limited to: smoking,
obesity, age, and medical conditions such as diabetes. Donor and
recipient site complications, as well as systemic complications need
to be considered, of which the most common are infection, vessel
thrombosis, hematoma or seroma formation, wound dehiscence,
and respiratory insufficiency.5
As the breadth of head and neck reconstruction has expanded,
the extirpative stage has largely remained the realm of otolaryngology, whereas the reconstruction stage has been shared by both
plastic surgery and otolaryngology. It is important to assure that
similar outcomes are obtained between these 2 reconstructive
specialties. If differences were to be found, it may serve to highlight
#
TABLE 1. List of Included Procedures

CPT Code
15756
15757
15758
20956
20957
20969
20970
20972
20973
49006
43496
Definition
Free muscle flap with or without skin graft with microvascular
anastomosis
Free skin flap with microvascular anastomosis
Free fascial flap with microvascular anastomosis
Bone graft with microvascular anastomosis; iliac crest
Bone graft with microvascular anastomosis; metatarsal
Free osteocutaneous flap with microvascular anastomosis;
other than iliac crest, metatarsal, or great toe
Free osteocutaneous flap with microvascular anastomosis; iliac crest
Free osteocutaneous flap with microvascular anastomosis; metatarsal
Free osteocutaneous flap with microvascular anastomosis;
great toe with web space
Free omental flap with microvascular anastomosis
Free jejunum transfer with microvascular anastomosis
CPT, current procedural terminology.
TABLE 2. List of Included Diagnoses

ICD-9 Code
140.xx
141.xx
142.xx
143.xx
144.xx
145.xx
146.xx
147.xx
148.xx
149.xx
150.xx
160.xx
161.xx
170.0x
170.1x
171.0x
172.0x
172.1x
172.2x
172.3x
172.4x
173.0x
173.1x
173.2x
173.3x
173.4x
210.xx
212.0x
213.0x
213.1x
230.0x
Definition
Malignant neoplasm of lip
Malignant neoplasm of tongue
Malignant neoplasm of major salivary glands
Malignant neoplasm of gum
Malignant neoplasm of floor of mouth
Malignant neoplasm of other and unspecified parts of mouth
Malignant neoplasm of oropharynx
Malignant neoplasm of nasopharynx
Malignant neoplasm of hypopharynx
Malignant neoplasm of other and ill-defined sites within the lip,
oral cavity, and phayrnx
Malignant neoplasm of esophagus
Malignant neoplasm of nasal cavities, middle ear,
and accessory sinuses
Malignant neoplasm of larynx
Malignant neoplasm of bone and articular cartilagebones of
skull and face, except mandible
Malignant neoplasm of bone and articular cartilagebones of
skull and face, mandible
Malignant neoplasm of connective tissue and other tissuehead,
face, and neck
Malignant melanoma of skinlip
Malignant melanoma of skineyelid, including canthus
Malignant melanoma of skinear and external auditory canal
Malignant melanoma of skinother and unspecified parts of face
Malignant melanoma of skinscalp and neck
Other malignant neoplasm of skin lip
Other malignant neoplasm of skineyelid, including canthus
Other malignant neoplasm of skinskin of ear and external
auditory canal
Other malignant neoplasm of skinskin of other and
unspecified parts of face
Other malignant neoplasm of skinscalp and neck
Benign neoplasm of lip, oral cavity, and pharynx
Benign neoplasm of respiratory and intrathroacic organsnasal
cavities, middle ear, and accessory sinuses
Benign neoplasm of bone and articular cartilagebones of skull
and face
Benign neoplasm of bone and articular cartilagelower jaw bone
Carcinoma in situ of digestive organslip, oral cavity, and pharynx
ICD-9, international classification of disease, revision 9.
e83
TABLE 3. Comparison of Outcomes Between Otolaryngology and Plastic Surgery

Occurrences (% of Reported)
Complication
Superficial SSI
Deep incisional SSI
Organ space SSI
Wound disruption
Pneumonia
Unplanned reintubation
DVT
Pulmonary embolus
Failure to wean from ventilator
Urinary tract infection
CVA
Cardiac arrest requiring CPR
Myocardial infarction
Sepsis
Septic shock
Return to OR
Flap failure
Perioperative transfusion
Death
Total
Otolaryngology
Plastic Surgery
Chi-squared P
38 (7.1%)
26 (4.8%)
6 (1.1%)
37 (6.9%)
38 (7.1%)
18 (3.3%)
6 (1.1%)
5 (1.0%)
43 (8.0%)
11 (2.0%)
3 (0.5%)
4 (0.7%)
7 (1.3%)
23 (4.3%)
3 (0.6%)
92 (17.1%)
22 (4.1%)
211 (39.2%)
6 (1.1%)
25 (4.6%)
13 (2.4%)
2 (0.4%)
17 (3.2%)
21 (3.9%)
7 (1.3%)
3 (0.6%)
2 (0.4%)
17 (3.2%)
6 (1.1%)
2 (0.4%)
0 (0%)
2 (0.4%)
12 (2.2%)
0 (0%)
52 (9.7%)
12 (2.2%)
123 (22.8%)
4 (0.7%)
13 (2.4%)
13 (2.4%)
4 (0.7%)
19 (3.5%)
17 (3.2%)
11 (2.0%)
3 (0.6%)
3 (0.6%)
26 (4.8%)
5 (0.9%)
1 (0.2%)
4 (0.7%)
5 (0.9%)
11 (2.0%)
3 (0.6%)
40 (7.4%)
10 (1.9%)
88 (16.3%)
2 (0.37%)
0.63
0.49
0.39
0.14
0.67
0.16
0.85
0.64
<0.05
0.88
0.96
0.05
0.22
0.65
0.1
0.44
0.77
0.15
0.92
Totals include 5 flaps performed by other services; statistically significant. CPR, cardiopulmonary resuscitation; CVA, cerebrovascular accident; DVT, deep venous thrombosis;
OR, operating room; SSI, surgical site infection.
areas of strengths and weaknesses between the 2 specialties that

could lead to improve training programs.
The goal of this study is to determine whether surgical specialty
training has any effect on the outcomes of patients undergoing
reconstruction of the head and neck via free tissue transfer. To
accomplish this, information provided by the American College of
Surgeons National Surgical Quality Improvement Program
(NSQIP) database was used. NSQIP began as a program mandated
by Congress in 1988 to assess the morbidity and mortality of
surgical cases in Veteran Affairs hospitals.6 Since then, it has
evolved into a vast, international database that collects clinical
data points from >250 participating hospitals for up to 30 days
postoperatively. It also analyzes, organizes, and provides this data
for the benefit of the participating hospitals with the purpose of
increasing the standard of surgical quality.7 It should be noted that if
differences between these 2 specialties were found based on this
database, it would be difficult to accurately extrapolate this to
discrepancies in training or surgical technique.
METHODS
The participant use files for the NSQIP database were analyzed
from 2005 to 2011. Initially all patients were identified that underwent a free tissue transfer based on Current Procedural Terminology
(CPT) codes (Table 1). This list of patients was further delimited
based on a primary diagnosis of a head or neck neoplasm (Table 2).
The determination of which service performed the reconstructive
free tissue transfer was made as follows. If the primary service was
listed as plastic surgery, the reconstruction was recorded as having
been performed by plastic surgery. If the primary service listed was
otolaryngology, a further examination was taken to determine if the
CPT code was listed as an other code or a concurrent code. By
definition an other CPT code listed for a patient was a procedure
performed by the primary service listed, as such the reconstruction
was recorded as having been performed by otolaryngology. The
definition of a concurrent CPT code was a procedure performed
e84
by a different surgical team and, for the purposes of this study, was
assumed to have been performed by plastic surgery and recorded as
such. Postoperative outcomes examined included superficial surgical site incisions (SSI), deep incisional SSIs, organ space SSIs,
wound disruptions, pneumonia, unplanned reintubations, pulmonary emboli, failure to wean from ventilator, urinary tract infections,
cerebrovascular accidents, cardiac arrests, myocardial infarctions,
blood transfusions, flap failure, deep venous thromboses, sepsis,
septic shock, and return to operating room. Chi-squared and t-test
statistical analyses were used to determine significance of interactions. A significance level of 5% was used.
RESULTS
During the time period examined, 539 free tissues transfers to the
head or neck were identified. Of these, 213 were performed
by plastic surgery, 321 were performed by otolaryngology, and
5 were performed by other services. The average patient age was
61.9 years. There were 371 males, 167 females, and 1 unlisted sex.
Intraoperative and postoperative complications are shown in Table 3.
There was a significantly increased rate of remaining on a
ventilator 48 hours postoperatively for flaps performed by plastic
surgery. Mean operative time for flaps performed by otolaryngology was 567 minutes versus 581 minutes for those performed by
plastic surgery (P 0.39). Plastic surgery performed significantly
more osseous free flaps than otolaryngology (57/213 [26.8%] for
plastic surgery versus 57/321 [17.8%] for otolaryngology;
P < 0.05). Total length of hospital stay was 11.2 and 13.0 days
for patients undergoing flaps performed by otolaryngology and
plastic surgery, respectively (P < 0.05). The mean hospital length of
stay for osseous flaps was 13.3 days and for soft tissue flaps was
11.5 days (P 0.06)
DISCUSSION
Our analysis of the data demonstrated that similar outcomes
are obtained between head and neck free tissue transfers performed
by otolaryngology and plastic surgery. Plastic surgery and
#
otolaryngology perform most head and neck free tissue transfers,

and our study set out to determine whether the differences in
surgical approach and methodology based on specialty training
have any effect on patients outcomes.
Patients having flaps performed by plastic surgery, on average,
spent 14 more minutes in the operating room, 581 as opposed to 567
minutes, and almost 2 more days in the hospital, 13 compared with
11.2 days. A longer hospital stay increases hospital costs and may
be associated with greater patient morbidity and mortality, but in
our study it did not correlate with a higher incidence of complications. Interestingly, more osseous flaps were performed by plastic
surgery. Of note, a relationship approaching significance was seen
between increased hospital length of stay and osseous flaps.
For the majority of postoperative complications recorded by
NSQIP, there was no statistical difference in outcomes. There were,
however, increased occurrences of remaining on a ventilator
48 hours postoperatively for flaps performed by plastic surgery,
which has the potential for complications such as airway trauma,
ventilator-associated pneumonia, and an increased need for sedation. This finding may represent the increased proportion of osseous
flaps performed by plastic surgery and the associated complexity
that these cases often portend.
Although the NSQIP database provided the basis for this study,
it has limitations that should be acknowledged. Primarily, the
database does not contain long-term outcomes, only collecting data
for 30 days postoperatively. As is the case in most reconstructive
procedures, technical differences during surgery can greatly affect
outcome, thus another limitation was the absence of descriptions of
surgical operative techniques. Lastly, there was no way of determining when or where surgical specialty training had been completed, which would provide a better understanding of the surgeons
performing these procedures. The results of this study are encouraging, as both specialties appear to receive satisfactory training in
microvascular reconstruction of head and neck defects. Institutional
practices will likely remain the driving force behind which specialty
performs the majority of these reconstructions.
CONCLUSIONS
Surgeon specialty training between plastic and otolaryngology
surgeons is not a significant predictor of 30-day postoperative
complication rates in head and neck free tissue transfers. The
few discrepancies that do exist between surgical specialty outcomes
are most likely multifactorial, with an optimal patient outcome
achieved through the correct processes of patient selection, surgical
technique, and postoperative management.
ACKNOWLEDGMENT
The American College of Surgeons National Surgical Quality
Improvement Program and the hospitals participating in the ACS
NSQIP are the source of the data used herein; they have not verified
and are not responsible for the statistical validity of the data
analysis or the conclusions derived by the authors.
6. Khuri SF, Henderson WG, Daley J, et al. Successful implementation of

the Department of Veterans Affairs National Surgical Quality
Improvement Program in the private sector: the Patient Safety in Surgery
study. Ann Surg 2008;248:329336
7. Cohen ME, Ko CY, Bilimoria KY, et al. Optimizing ACS NSQIP
modeling for evaluation of surgical quality and risk: patient risk
adjustment, procedure mix adjustment, shrinkage adjustment, and
surgical focus. J Am Coll Surg 2013;26:
Assessment of Hematological
Factors Involved in Development
and Prognosis of Idiopathic
Sudden Sensorineural
Hearing Loss
Kasim Durmus, MD, Hatice Terzi, MD,y
Tuba Dogan Karatas, MD, Mansur Dogan, MD,
Ismail Onder Uysal, MD, Mehmet Sencan, MD,y
and Emine Elif Altuntas, MD
Objective: The aim of this study was to investigate the possible
effects of routine hematological parameters on the development and
prognosis of idiopathic sudden sensorineural hearing loss in patients
applying to our clinic.
Study design: A retrospective clinical study.
Setting One academic health center from 2008 to 2014.
Patients and intervention: One hundred forty patients with sudden
hearing loss and 132 healthy controls were included in the present
study.
Results: Patients having idiopathic sudden sensorineural hearing
loss were divided into 2 subgroups based on whether they recovered
(complete, partial, and slight recovery) (Group 1; n 83, 59.3%) or
not (Group 2; n 57, 40.7%) during the follow-up term. Group 1,
Group 2, and the controls differed statistically significantly in terms
of neutrophil-to-lymphocyte ratio (P 0.001), platelet-to-lymphocyte ratio (P 0.001), lymphocytes % (P 0.001), mean corpuscular
hemoglobin (P 0.019), mean corpuscular hemoglobin concentration (P 0.015), platelet (P 0.001), mean platelet volume
(P 0.001), platelet distribution width (P 0.009), and glucose
(P 0.001). The study groups and the controls did not have any
significant difference in terms of other laboratory parameters affecting the prognosis of Idiopathic sudden sensorineural hearing loss.
Conclusions: The results the authors obtained showed that laboratory parameters such as lymphocyte, lymphocyte%, platelet,
REFERENCES
1. Hurvitz KA, Kobayashi M, Evans GR. Current options in head and neck
reconstruction. Plast Reconstr Surg 2006;118:122e133e
2. Davis JS. Address of the president: the story of plastic surgery. Ann Surg
1941;113:641656
3. Tamai S. History of microsurgery. Plast Reconstr Surg 2009;124
(6 suppl):e282e294
4. Wong CH, Wei FC. Microsurgical free flap in head and neck
reconstruction. Head Neck 2010;32:12361245
5. Pohlenz P, Klatt J, Schon G, et al. Microvascular free flaps in head and
neck surgery: complications and outcome of 1000 flaps. Int J Oral
#
From the Department of Otolaryngology; and yDepartment of Internal

Medicine, Faculty of Medicine, Cumhuriyet University, Sivas, Turkey.
Received February 12, 2015.
Address correspondence and reprint requests to Kasim Durmus, MD,
Department of Otolaryngology, Faculty of Medicine, Cumhuriyet
University, 58140 Sivas, Turkey; E-mail: kasimdurmus58@gmail.com
ISSN: 1049-2275
DOI: 10.1097/SCS.0000000000002241
e85
mean platelet volume, platelet distribution width, neutrophil-tolymphocyte ratio, platelet-to-lymphocyte ratio, mean corpuscular
hemoglobin, and mean corpuscular hemoglobin concentration may
be indicative for prognosis and treatment success in groups of
patients suffering idiopathic sudden sensorineural hearing loss in
whose etiology many factors play a role.
Key Words: Laboratory parameters, outcome, prognostic marker,
sudden hearing loss
hearing loss of more than 30 dB at 3 consecutive frequencies

occurring within 3 days is defined as idiopathic sudden sensorineural hearing loss (ISSHL) which is a common medical emergency.1 Although most of the patients having ISSHL are considered
to be idiopathic, the known causes of the disease are viral infection
of the labyrinth or cochlear nerve, bacterial and protozoan infections, vascular incident, vascular occlusion, labyrinthine membrane
rupture, perilymphatic hypoxia inflammatory, ototoxicity drugs,
neoplastic, metabolic conditions, and autoimmune disorders.2
Blood platelets are the smallest cells of the peripheral blood and
they are involved in hemostasis. Considered as an important source
of prothrombotic agents associated with inflammatory markers,
platelets are also involved in the initiation and propagation of
vascular and inflammatory diseases.3 They also play a very significant role in the formation of thrombosis in the vessel. Mediators
and substance that are crucial for coagulation, inflammation,
thrombosis, and atherosclerosis are released by platelets.4,5 Platelets
are heterogeneous in volume and density. In evaluating the size of
thrombocytes, the volume of platelets is used as an objective
parameter. Large platelets have a higher metabolic and enzymatic
activity and thrombotic potential.6,7 In terms of tendency for
aggregation, large platelets have a higher tendency when compared
with small platelets. Platelet production rate and stimulations are
reflected by the mean platelet volume (MPV) that is one of the
platelet function indices.8 MPV level is increased in vascular events
such as thromboembolism, venous and arterial thrombosis, atherosclerosis, or acute syndromes.911 The correlation between vascular
ischemic events and MPV has been studied in many studies aiming
to establish whether changes in platelet volume markers are
prophylactically and diagnostically important in thrombotic and
prothrombotic cases.12
Higher platelet counts may reflect increased thrombocyte activation, which has pivotal role for megakaryocytic proliferation and
produce relative thrombocytosis. In clinical practice, neutrophil
lymphocyte ratio (NLR) and plateletlymphocyte ratio (PLR) are
used to evaluate systemic inflammation.13 NLR, which has been
introduced recently, is a simple and cost-effective predictor. Many
studies have shown that NLR is increased in atherosclerosis,
cardiovascular, and cerebrovascular diseases such as hypertension,
unstable angina pectoris, myocardial infarction, and stroke.1417
As a result, in recent years, there are many studies evaluating the
effects of various hematological parameters on the prognosis
ISSHL.12,1821 The aim of this study was to investigate the possible
effects of routine hematological parameters on the development and
prognosis of ISSHL.
METHODS
Study Population
One hundred sixty consecutive patients having idiopathic sudden sensorineural hearing loss (ISSHL) and treated at our department between 2008 and 2014 loss were reviewed retrospectively.
The study group consisted of 140 patients having ISSHL and
e86
consenting to participate in the study and 132 controls having no

ear pathology. Having a hearing loss of at least 30 dB in 3 consecutive frequencies in 72 hours was defined as ISSHL.1 In order to
compare the biomarker profile, 132 age and sex-matched healthy
individuals having no hearing loss at regular health check-ups at our
hospital were included in the study too. None of these individuals
had an acute inflammation or otologic disease.
Inclusion criteria were applying to hospital within 1 week from
the onset of the disease, receiving no previous steroid treatment,
presence of blood sample, and undergoing pure-tone hearing test
during the first visit. Those having an acute inflammation, infection,
a history of otologic surgery, trauma, or barotrauma during the
previous 4 weeks, having cerebellopontine angle pathology or
congenital cochlear malformations, neurologic disorders predisposing to hearing loss, recent use of ototoxic medications, having
neoplasm within the previous 2 years, or having other major
diseases (such as heart failure, hypertension, coronary artery disease, cor pulmonale, liver or renal dysfunction, diabetes mellitus,
chronic obstructive pulmonary disease, obstructive sleep apnea,
connective tissue diseases, and inflammatory bowel diseases) and
any otologic disease such as otitis media during the last 4 weeks,
having chronic otitis media, otosclerosis and Menieres disease
were excluded from the study.
After the diagnosis of ISSHL, corticosteroid treatment was
initiated and serum samples were obtained. All the patients were
given IV metilprednisolon (1 mg/kg/day Prednol-L ampoule,
Mustafa Nevzat Drug Industry, Istanbul, Turkey), with a
progressive dose reduction by 10 mg per 2 days maintained for
at least 2 weeks. While on the corticosteroids, the patients were
given the H2 receptor inhibitor ranitidine 1 1 ampoule i.v.
(Ulcuran ampoules 50 mg/2 mL IV, Yavuz Drug Industry, Turkey),
oral vitamin B1 (2 250 mg thiamine hydrochloride), and B6
(250 mg pyridoxine hydrochloride; Nerox B tablet, Abdi Ibrahim
Pharmaceutical Company, Istanbul, Turkey) for a 3-month term.
Then, 100 mg pentoxfylline (Vasoplan AMP 100 mg/5 mL Mustafa
Nevzat Drug Industry, Istanbul, Turkey) was added into 500 mL
Voluven (Fresenius Kabi Drug Industry, Frankfurt Am Main,
Germany) and administered by intravenous infusion. The dose
of pentoxfylline was added every other day and the treatment
lasted 8 days.
A general physical examination and an audiological evaluation
were carried out and blood parameters were studied in all the
patients.
An Ethical Committee approval was obtained and the study was
conducted in accordance with the Helsinki Declaration. Informed
consent was obtained from all the participants.
Audiological Evaluation
Hearing data of all 140 patients obtained by AC-40 Interacoustics Clinic Audiometer (Interacoustics, Assen, Denmark) at baseline
and after the treatment (at the end of the 4th week) were gathered
from the audiological evaluation form of each patient.
All of the ISSHL patients underwent a standard evaluation that
consisted of a pure-tone speech audiometry. Pure-tone thresholds
were obtained for air conduction at 250, 500, 1, 2, 4, and 6 and for
bone conduction at 250, 500, 1, 2, and 4 kHz, respectively. Audiologic data were reported using the methods recommended by the
Hearing Committee of the American Academy of Otolaryngology
Head and Neck Surgery. On the basis of Siegel criteria,22,23 a
classification was made in accordance with the treatment success
and average pure-tone averages observed during the follow-ups
after a 1-month term. Patients having idiopathic sudden sensorineural hearing loss were divided into 2 subgroups by taking into
account whether their pure-tone averages (PTA) recovered
#
TABLE 1. Siegels Criteria of Hearing Improvement

Group
Hearing Recovery
Complete
recovery
Partial
recovery
Slight
recovery
No improvement
II
III
IV
Definition
Patients having a final hearing level better than
25 dB regardless of the size of the gain
Patients having a gain more than 15 dB and having a
final hearing level between 25 and 45 dB
Patients having a gain more than 15 dB and having a
final hearing level poorer than 45 dB
Patients having again less than 15 dB
(complete, partial, and slight recovery) (Group 1; n 83, 59.3%) or

not (Group 2; n 57, 40.7%). Hearing data of all 140 patients
obtained by AC-40 Interacoustics Clinic Audiometer (Interacoustics) at baseline and after the treatment (at the end of the 4th week)
were gathered from the audiological evaluation form of each
patient (Table 1).
Hematologic Examinations
Blood samples were tested for all patients at the first visit to
prevent adverse effects of the steroid treatment in high-risk patients
like those having DM, CBC, and routine chemistry. Biochemical
analysis and hemogram were performed using the peripheral venous
blood samples obtained at admission. Blood samples were collected
into tubes containing calcium EDTA. A fully automated blood cell
counter (Mindray BC-6800) was used for CBC measurements. All
samples were run in duplicate, and the mean values were used for
statistical analysis. NLR was the ratio of absolute neutrophil to
lymphocyte count, while PLR was the ratio absolute platelet
to lymphocyte count.
The Statistical Package of Social Science (SPSS Inc, Chicago,
IL) for Windows version 14.0 was used to analyze the data.
In evaluating the data, paired-sample t test was used when the
parametric test assumptions were met and MannWhitney U and
Wilcoxon tests were used when the said parameters were not met.
0.05 was regarded as significant.
RESULTS
The mean (SD) age of the patients with ISSHL and the control group
was 47.43 16.14 (maxmin: 8714 years) and 44.42 16.22
(maxmin: 8416 years) years, respectively. In total, 37.1%
(n 52) of the ISSHL group and 38.6% (n 51) of the control

group were women, while 62.9% (n 88) of the ISSHL group and
61.4% (n 81) of the control group were men. The groups were
similar in terms of age and sex (P 0.126, P 0.703).
In the ISSHL group, hearing loss was unilateral in all the patients
(right ear 41.43% and left ear 58.57%). The distribution of patients
based on the recovery of hearing ability in accordance with the
Siegel classification was as follows: 50% (n 70) achieved complete recovery, 5% (n 7) showed partial recovery, 4.3% (n 6)
showed slight recovery, and 40.7% (n 57) showed no improvement. ISSHL patients were divided into 2 subgroups based on
whether they recovered (complete, partial, and slight recovery)
(Group 1; n 83, 59.3%) or not (Group 2; n 57, 40.7%) during the
follow-up term.
In ISSHL patients, the mean PTA was 43.65 25.86 (maxmin:
11810 dB) dB just before the treatment. On the 14th day of the
treatment, the mean PTA of the ISSHL patients was 31.11 28.78
(maxmin: 1183 dB) dB. In Group 1, the mean PTA was
40.42 22.84 dB just before the treatment and 20.02 18.79 dB
at the end of the treatment (t 12.26, P 0.001). In Group 2,
the mean PTA was 48.36 29.29 dB just before the treatment
and 47.26 33.03 dB at the end of the treatment (t 0.63,
P 0.526).
When the hearing threshold obtained by pure-tone audiometric
evaluation performed at the time of ISSHL diagnosis was compared, the groups did not show any statistically significant difference at low frequencies (250, 500, and 1000 Hz). However, the
hearing thresholds of the groups between 2 and 6 kHz were significantly different (P < 0.005). When the hearing thresholds of the
groups at the end of the treatment were compared, the difference at
all frequencies was found to be statistically significant (P < 0.005).
Table 2 shows that the degree of hearing loss was comparable in
2 groups.
With respect to the neutrophil-to-lymphocyte ratio (P 0
.001), platelet-to-lymphocyte ratio (P 0 .001), lymphocytes
% (P 0.001), mean corpuscular hemoglobin (P 0.019), mean
corpuscular hemoglobin concentration (P 0.015), platelet
(P 0.001), mean platelet volume (P 0.001), platelet distribution width (P 0.009), and glucose (P 0.001), Group 1,
Group 2, and the controls showed statistically significant differences (Table 3) in group values, but large overlaps in ratios
prevented segregating a cutoff value to segregate good from poor
prognosis using these statistics by themselves. The study groups
and the controls did not show any significant difference in terms
of the other laboratory parameters affecting the prognosis of
ISSHL. Routine laboratory parameters showing a statistically
significant difference between Group 1 and Group 2 are shown
in Table 4.
TABLE 2. Pure-Tone Hearing Thresholds of ISSHL Patients Before and After Treatment
Initial
PTA
250 (Hz)
500 (Hz)
1000 (Hz)
2000 (Hz)
4000 (Hz)
6000 (Hz)
Follow-Up
Group 1
Group 2
Results
Group 1
Group 2
Results
54.55 20.99
54.01 21.22
49.67 23.98
44.01 25.05
51.54 25.24
55.81 25.63
56.61 28.09
55.39 28.14
57.40 26.71
54.77 27.43
62.14 28.32
67.66 26.69
0.621
0.743
0.076

0.018

0.022

0.009
28.36 18.56
23.18 17.02
21.49 17.50
21.97 21.50
30.10 23.41
34.27 27.42
53.98 28.18
53.24 28.55
54.82 27.79
50.52 27.65
64.29 29.49
66.22 28.17
0.001

0.001

0.001

0.001

0.001

0.001
The data are expressed in mean standard deviation or in numbers and percentages; PTA, pure-tone average.

P < 0.05 value was regarded as significant while the significant differences between the groups are shown in bold.
e87
TABLE 3. The Demographic and Clinical Characteristics of the Study Populations (Patients and Controls)
Patients Group (N 140)
Variables
Control Group (N 132)
Group 1 (N 83)
Group 2 (N 57)
P Value
44.42 16.22
47.02 15.72
48.03 16.85
0.292
32
100
1.80 0.47
114.55 26.57
7.02 1.28
63.64 61.58
32.83 6.05
5.98 1.16
2.20 1.38
0.39 0.19
4.12 0.86
2.32 0.45
0.43 0.10
0.15 0.95
0.26 0.14
16.13 12.75
5.58 3.99
43.80 4.66
86.50 3,26
29.55 1.31
34.04 0.75
13.29 0.57
258.59 50.63
9.12 0.84
0.23 0.05
16.06 1.99
90.48 5.72
21.03 4.25
0.63 0.12
24.07 5.70
19.34 4.59
53
30
2.83 3.04
101.97 67.28
8.93 8.13
63.22 10.18
28.98 8.51
5.63 1.92
1.89 1.74
0.37 0.24
5.21 2.44
2.23 0.78
0.44 0.21
0.14 0.12
0.27 0.21
14.63 1.64
5.14 1.37
43.43 4.42
86.73 4.36
29.21 1.75
33.67 1.03
13.28 0.83
191.12 34.88
8.19 1.28
0.41 1.62
16.73 5.45
114.71 70.77
16.49 11.11
1.00 1.12
27.64 24.60
23.52 12.34
35
22
3.75 4.06
165.79 58.19
7.94 2.19
66.06 9.16
25.70 7.68
5.89 1.57
2.05 1.59
0.37 0.23
6.20 6.16
1.84 0.55
0.45 0.15
0.15 0.11
0.24 0.17
14.47 1.87
5.03 0.63
42.83 4.86
85.30 4.47
28.83 2.05
33.78 1,25
13.41 0.96
282.51 61.11
10.14 1.66
0.24 0.06
18.35 7.25
148.28 76.17
17.85 13.31
1.17 1.37
25.36 16.71
23.40 13.49
0.001

0.001
0.864
0.074

0.001
0.241
0.446
0.729
0.138

0.001
0.175
0.315
0.676
0.493
0.383
0.417
0.078

0.019

0.015
0.538

0.001

0.001
0.313

0.009

0.001
0.801
0.869
0.922
0.830
Age, yr
Sex (n)
Male
Female
NLR
PLR
WBC, 103/u
Neutrophil, %
Lymphocytes, %
Monocytes, %
Eosinophils, %
Basophil, %
Neutrophil, 103/u
Lymphocytes, 103/u
Monocytes, 103/u
Eosinophils, 103/u
Basophil, 103/u
Hemoglobin, g/dL
RBC, 106/u
HCT, %
MCV, fL
MCH, pg
MCHC, g/dL
RDW, %
PLT, 103/u
MPV, fL
PCT, %
PDW, %
Glucose, mg/dL
BUN, mg/dL
Cre, mg/dL
ALT, mg/dL
AST, mg/dL
ALT, alanine transaminase; AST, aspartate transaminase; BUN, blood urea nitrogen; Cre, creatinine; HCT, hematocrit; MCH, mean corpuscular hemoglobin; MCHC, mean
corpuscular hemoglobin concentration; MCV, mean corpuscular volume; MPV, mean platelet volume; NLR, neutrophil-to-lymphocyte ratio; PCT, platelet crit; PDW, platelet
distribution width; PLR, platelet-to-lymphocyte ratio; PLT, platelet; RBC, red blood cell; RDW, red cell distribution width; WBC, white blood cell. The data are expressed in
mean standard deviation or in numbers and percentages.

P < 0.05 value was regarded as significant while the significant differences between the groups are shown in bold.
DISCUSSION
TABLE 4. Comparisons of ISSHL Patients With and Without Improvement
Following Standard Treatment Protocol
Lymphocytes %
Lymphocytes, 103/u
PLT, 103/u
MPV, fL
PDW, %
PLR
NLR
Glucose, mg/dL
Group 1
Group 2
Result
28.98 8.51
2.23 0.78
191.12 34.88
8.19 1.28
16.73 5.45
101.97 67.28
2.83 3.04
114.71 70.77
25.70 7.68
1.84 0.55
282.51 61.11
10.14 1.66
18.35 7.25
165.79 58.19
3.75 4.06
148.28 76.17
0.015

0.001

0.001

0.001

0.001

0.001

0.001

0.001
MPV, mean platelet volume; NLR, neutrophil-to-lymphocyte ratio; PDW, platelet

distribution width; PLR, platelet-to-lymphocyte ratio; PLT, platelet. The data are
expressed in means standard deviation or in numbers and percentages.

P < 0.05 value was regarded as significant while the significant differences
between the groups are shown in bold.
e88
The aim of this study was to investigate the effects of routine

hematological parameters on the development and prognosis of
ISSHL. We analyzed 140 patients having unilateral ISSHL and
treated with a standardized treatment protocol during the previous
6 years. The most important finding of our study was that NLR,
PLR, platelet count, mean platelet volume (MPV), and platelet
distribution width (PDW) levels were significantly higher and
lymphocytes and lymphocytes levels were significantly lower in
patients with ISSHL when compared with the control group.
Observing significantly higher NLR, PLR, PDW, MPV, and PLT
levels and significantly lower lymphocytes and lymphocytes %
levels in Group 2 when compared with Group 1 was another
important finding of our study.
ISSHL is a frequently encountered medical condition in ENT
practice. It is characterized by sudden onset hearing loss, which may
become evident within hours or days.24 Various conditions such as
bacterial, viral, and protozoan infections, vascular occlusion, mechanisms associated with the immune system, ototoxicity drugs
#
TABLE 5. In Patients With Idiopathic Sudden Sensorineural Hearing Loss, NLR Value of Published Studies Between the 2013 and 2014 Years and Results of Current
Study
Study References
Study Design
Population Studied (n)
Findings
Ulu et al (2013)
Clinical study
ISSHL patients (47) The control group was composed

of age and sex-matched healthy subjects (45)
Seo et al (2014)
Clinical study
ISSHL patients (1046)
Ozler et al (2014)
Clinical study
ISSHL patients (40) The control group was composed

of age and sex-matched healthy subjects (40)
Durmus et al
(2015)
Clinical study
ISSHL patients (140) The control group was

composed of age and sex-matched healthy
subjects (132)
NLR 3.96 2.95 in ISSHL patients. NLR 1.82 0.79 in control patients.
(P < 0.001)
NLR 2.81 2.19 in recovered ISSHL patients. NLR 4.88 3.20 in
unrecovered ISSHL patients (P 0.001)
NLR 3.50 3.38 in recovered ISSHL patients NLR 5.98 4.22 in
unrecovered ISSHL patients (P < 0.001)
NLR 5.53 1.72 in ISSHL patients. NLR 2.73 0.81 in control patients.
(P 0.001)
Hearing loss was classified as mild (<40 dB loss for any frequency, Group A),
moderate (up to 80 dB, Group B), and severe (profound, >80 dB, Group C).
NLR 5.53 1.72 in Group A patients. NLR 5.29 1.81 in Group B
patients. NLR 5.82 1.72 in Group C patients.
NLR 2.83 3.04 in Group 1 (complete, partial, and slight recovery) ISSHL
patients. NLR 3.75 4.06 in Group 2 (unrecovered) ISSHL patients.
NLR 1.80 0.47 in control patients. (P 0.001)
(salicylates, amino-glycosides) and traumatic, vascular, neoplastic,

and metabolic conditions may cause ISSHL. However, the etiology
of the disease is still unknown. Infectious, immunologic and
inflammatory reasons, and microcirculatory failure are among those
hypothesized to be associated with ISSHL. It could also be associated with an alteration in ear microcirculation due to genetic
prothrombotic susceptibility or cardiovascular risk factors such
as hypertension and diabetes.25,26 A high blood viscosity may also
damage the ear microcirculation and cause hearing loss.27,28
Complete blood count is a routine, inexpensive, and easy
method that yields information on the red and white cells, the
platelets, the count, and dimensions of subgroups of cells including red cell distribution width, platelet distribution width, and
parameters like the platelet lymphocyte ratio, neutrophil
lymphocyte ratio.13
NLR, PLR, and MPV have been defined as novel potential
markers in determining inflammation in multiple diseases including
oncological, cardiological, and cerebrovascular diseases, end-stage
renal disease, and inflammatory conditions such as ulcerative
colitis, ankylosing spondylitis, pelvic inflammatory disease, and
appendicitis.2932
In early diagnosis of thromboembolic diseases, platelet indices
are potential useful markers too. MPV and PDW measure the
variation in platelet size and are markers of platelet activation.33 36 As platelet activation caused morphological changes
in platelets, several investigators have used some platelet indices
measured by hematology analyzers. MPV is one of the most
extensively studied platelet activation markers. Novel platelet
indices such as mean platelet component (MPC) and PDW are
among the recently studied activation markers.3739
As a result, there are many studies in the literature showing an
association between platelet volume and coagulation.40 43 All of
these studies point out that there may be an association between
MPV and diseases of thromboembolic origin. From this point of
view, Karli et al12 studied whether MPV was a predictive parameter
for ISSHL in 46 ISSHL patients, and found that it was not a
predictive parameter in the diagnosis of MPV. Moreover, they
stated that there was no difference when ISSHL patients and the
control group were compared in terms of platelet values.
Karli et al12 stated that cochlear ischemia could have a role in the
etiology of ISSHL but neither their own clinical observations nor
some clinical and experimental studies supported the role of
vascular risk factors in the etiology of ISSHL. Contrary to the
study of Karli et al,12 when we compared the 3 groups in terms of
MPV values, we found a significant difference. Moreover, MPV
#
values were the lowest in the recovering group 1, while group 2 had
statistically significantly higher MPV values when compared with
group 1 and the controls. This suggested us that MPV could be an
important prognostic parameter of MPV in ISSHL patients.
In a study conducted by Kanzaki et al19 on ISHHL patients in
2014, it was found that there could be an association between high
fibrinogen levels and bad prognosis of the disease.
Witting et al21 studied whether patients with ISSHL and its
comorbidity also influence routine pretherapeutic laboratory values
and whether these values have prognostic influence on hearing
recovery after a standardized combined glucocorticoid and rheological therapeutic regime. They found that hyperfibrinogenemia
seemed to be a risk factor for ISSHL. They, therefore, stated that
hyperfibrinogenemia was not only a risk factor for ISSHL but also a
positive prognostic marker of outcome when using a rheological
regime to treat ISSHL.
In a case-control study, Weiss et al20 found elevated fibrinogen
concentrations and a higher prevalence of T allele carriers of the
glycoprotein (Gp) Ia C807T polymorphism in ISSHL patients.
Their findings suggested that ISSHL had a vascular/rheological
origin with features unique to thrombosis in the inner ear artery that
may include complex interrelations among platelet glycoproteins
and plasma fibrinogen.
Seo et al44 showed that ISSHL patients had significantly higher
NLR and PLR values and stated that NLR level could be taken into
account as a novel potential marker to predict the prognosis in terms
of recovery. Seo et al44 found that the mean NLR was 4.48 3.92
and mean PLR was 169.25 102.88 in the patient group. Both
mean NLR and PLR values were higher in the patient groups when
compared with the controls (both P < 0.001). The optimal cut-off
values have been suggested to be >3.95 and >150.00 for NLR and
PLR, respectively.45 In our study, the PLR values of groups 1, 2 and
control were 101.97 61.28, 165.79 59.19, and 114.55 26.57,
NLR values of groups 1, 2 and control were 2.83 3.04,
3.75 4.06, and 1.80 0.47, respectively.
Similarly, in studies conducted by Ozler46 and Ulu et al,47 NLR
levels of the patients were higher when compared with the control
group. NLR levels in ISSHL patients in various studies and results
of our study are summarized in Table 5. In our study, we found that
groups 1 and 2 in aggregate had higher NLR levels when compared
with the controls but that no single specific value separated these
2 groups. Moreover, when groups 1 and 2 were compared, patients
in recovering group 1 had lower NLR values when compared with
group 2. For this reason, we think that NLR value can be a potential
marker for prognosis in such patients. When PLR values were
e89
compared, group 1 had lower PLR values when compared with the
control group while group 2 had higher values. Therefore, we think
that it would be appropriate to regard PLR levels as a reliable
indicator of prognosis too. However, according to the available data
in the literature and this study, it is not appropriate to determine cutoff value of this test. Further studies aiming to determine cut-off
value of NLR and PLR values will be helpful to draw reliable
conclusions.
Kassner et al48 found that a significant increase in proinflammatory CD40, TNF-a, cyclooxygenase-2, or CD38-positive T or B
lymphocytes values in ISSHL patients. Their data suggested an
enhanced extravasation of proadhesive and proinflammatory
lymphocytes from the peripheral circulation, which may contribute
to ISSHL disease induction as well as progression and, thus, may be
proposed as a novel therapeutical target. In our study, Lymphocytes
% and lymphocytes levels were statistically significantly lower in
ISSHL patients when compared with the control group in aggregate
but no differentiating cutoff value was identifiable. The difference
between groups 1 and 2 was statistically significant, while both of
the values were higher in group 1 when compared with group 2.
This showed the importance of relatively low lymphocyte due to
neutrophil predominance and thus the importance of inflammation
in the etiology of the disease. Receiving a better response to
treatment in the group having lower lymphocyte levels is evidence
to this. We believe that the difference between the recovering
(Group 1) and nonrecovering (Group 2) in terms of percentage
and count of lymphocyte is an important marker in following both
the prognosis and the treatment response.
The aim of the erythrocyte indices is to provide estimates of the
average size of circulating eythrocytes (mean corpuscular volume
(MCV)), the average concentration of hemoglobin per erythrocyte
(mean corpuscular hemoglobin concentration (MCHC)), and the
average quantity of hemoglobin within erythrocytes (mean corpuscular hemoglobin (MCH)). The MCHC is a measure of the concentration of hemoglobin in an average circulating erythrocyte.49
The main function of erythrocytes is gas exchange. Hemoglobins in
its cytoplasm play the main role in oxygen transportation. MCH and
MCHC are the hematologic parameters showing the hemoglobin
content of erythrocytes. Our literature review did not yield any
studies evaluating erythrocyte indices in ISSHL patients. In our
study, hemoglobin value was 16.13 12.75 in the control group,
14.63 1.64 in group 1, and 14.47 1.87 in group 2. The difference
was not significant. However, MCH and MCHC values were found
to be lower in ISSHL patients when compared with the controls
[MCH was 29.55 1.31 in the control group, 29.21 1.75 in group
1, and 28.83 2.05 in group 2 (P 0.019). MCHC was
34.04 0.75 in the control group, 33.67 1.03 in group 1, and
33.78 1.25 in group 2 (P 0.015)]. The MCH and MCHC may be
a significant difference between controls and ISSHL, but no cut-off
value is identifiable.
Hyperglycemia is not identified as a significant prognostic
factor for idiopathic sudden sensorineural hearing loss in any
literature. Therefore, Ryu et al50 investigated the prognostic value
of hyperglycemia in predicting hearing recovery. And they suggest
that hyperglycemia may be a potential negative prognostic factor
for hearing recovery in idiopathic sudden sensorineural hearing
loss. Yasan et al18 investigated the possible effects of routine blood
chemistry and hematological parameters on the development and
prognosis of disease in patients with ISSHL. And they found that a
statistically significant difference between the patients of ISSHL
and those of control subjects with respect to the fasting blood
glucose (P 0.030). In our study, glucose level was statistically
significantly higher in ISSHL patients when compared with the
control group. Moreover, when groups 1 and 2 were compared,
patients in recovering group 1 had lower glucose values when
e90
compared with group 2. For this reason, we think that glucose value
can be a potential marker for prognosis in such patients.
When ISSHL groups were compared, there was no statistically
significant difference in terms of MCH, MCHC, and hemoglobin
values. For this reason, although MCH and MCHC are not suitable
markers in terms of prognosis, we believe that they can be used as
novel markers in determining the etiology in ISSHL patients as they
draw attention to ischemic risk factors.
CONCLUSIONS
The results we obtained showed that laboratory parameters used
routinely in clinical practice to vary too much to allow them to be
used to predict prognosis in ISSHL patients but, in aggregate, the
PLR and the NLR are statistically significantly higher in poor
prognosis patients. Our results comply with most of studies published in the literature in recent years, and, contrary to the results of
Karli et al,12 show that MPV can be an important marker in terms of
prognosis and treatment success in ISSHL patients. Moreover,
similar to the results obtained by Kassner et al,48 we believe that
lymphocyte and lymphocyte % values can be used as easy and costeffective markers used routinely in clinical practice vary too much
to allow them to be used to predict prognosis in ISSHL patients but,
in aggregate, the lymphocyte and lymphocyte % values are statistically significantly lower in poor prognosis patients.
In our study, hemoglobin values of the patient groups were lower
than those of the controls; however, the difference was not statistically significant. MCH and MCHC, the erythrocyte indices,
were lower in the control group when compared with ISSHL
patients. Our literature review did not yield any studies on this
issue. Although these 2 parameters are not suitable markers in terms
of prognosis, we believe that they can be used as novel markers in
determining the etiology in ISSHL patients as they draw attention to
ischemic risk factors.
The results we obtained suggested that PLR and NLR could be
associated with the prognosis in patients having ISSHL. While the
PLR and NLR in group aggregated data are associated with
the prognosis in ISSHL, our data do not allow us to determine a
reliable differentiating cut-off value for these ratios.
In conclusion, PLR, PDW, NLR, PLT, MPV, lymphocytes %,
and lymphocytes level can be taken into consideration as potential
novel markers in predicting prognosis in terms of recovery but
further studies are needed to ascertain how to use these markers to
provide sufficiently accurate segregation of prognosis in ISSHL
patients.
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Sport-Related Maxillofacial
Fractures
Muhammad Ruslin, DDS, MS,y Paolo Boffano, MD,y
Y.J.D. ten Brincke, BSc,y Tymour Forouzanfar, DDS, PhD,y
and Henk S. Brand, PhDyz
Abstract: Sports and exercise are important causes of maxillofacial
injuries. Different types of sports might differ in frequency and type
e91
of fractures. The aim of the present study was to explore the possible
relation between the types of sport practiced and the frequency and
nature of the facial bone fractures of patients presenting in an oral
and maxillofacial surgery department of a Dutch university center.
This study is based on an analysis of patient records containing
maxillofacial fractures sustained between January 1, 2000 and April
1, 2014 at the Vrije Universiteit University Medical Center (VUmc)
in Amsterdam, The Netherlands. The present study comprised data
from 108 patients with 128 maxillofacial fractures. Seventy-nine
percent of the patients were male and 21% were female. The
patients ranged in age from 10 to 64 years old with a mean age
of 30.6 12.0. The highest incidence of sport-related maxillofacial
fractures occurred in individuals between the ages of 20 and 29. The
most common sport-related fractures were zygoma complex fractures, followed by mandible fractures. Soccer and hockey were the
most prominent causes of sport-related maxillofacial trauma in the
present study. Coronoid process fractures were only observed in
soccer players and not in other sports groups. Mandible angle
fractures were relatively more frequent in rugby than in other
sports. The results of this study suggest a relation between type
of sport and the nature and frequency of the fractures it causes.
Key Words: Maxillofacial, fractures, sport
ajor causes of maxillofacial injuries are traffic accidents,

falling, and (domestic) violence. Sports and exercise are
also important causes of maxillofacial injuries. Sports cause
approximately 5% of all mandible fractures and 9% of the
fractures in the upper two-thirds of the face. Sport-related
accidents are also responsible for approximately 10% of all
midfacial.1 4
Elhammali et al5 found in their study on sport-related injuries a
significant prevalence of the mid-facial complex (67%) followed by
the mandible (29%) and skull base (4%).5 In their review study
concerning sports-related maxillofacial trauma, Kunamoto et al6
suggested that different types of sports differ in frequency and type
of fractures. In Italy, soccer is the main cause of maxillofacial
trauma, with frequent fractures of the zygomatic bone (44%), the
nasal bone (29%), and the mandible (15%). A previous, larger study
in Italy also found the same 3 types of fractures most commonly in
soccer players, but in different orders: nasal bone fractures (62%),
zygomatic bone fractures (15%), and mandible fractures (11%). In a
study performed in Brazil, the investigators found that the majority
of soccer-related fractures consisted of nasal bone (35%) and
orbito-zygomatic complex (35%) followed by mandible (16%),
From the Department of Oral and Maxillofacial Surgery, Faculty of
Dentistry, University of Hasanuddin, Makassar, Indonesia; yDepartment
of Oral and Maxillofacial Surgery/Oral Pathology, VU University
Medical Center/Academic Center for Dentistry Amsterdam (ACTA);
and zDepartment of Medical-Dental Interaction, Academic Center for
Dentistry Amsterdam (ACTA), Amsterdam, The Netherlands.
Received September 15, 2014.
Address correspondence and reprint requests to Muhammad Ruslin, DDS,
MS, Department of Oral and Maxillofacial Surgery/Oral Pathology, VU
University Medical Center, P.O. Box 7057, 1007 MB, Amsterdam, De
Boelelaan 1117 1081 HV Amsterdam, The Netherlands;
E-mail: m.ruslin@vumc.nl; ruslin_oms@yahoo.com
ISSN: 1049-2275
DOI: 10.1097/SCS.0000000000002242
e92
orbital region (13%), frontal bone (2%), and nasoorbitoethmoid

complex (2%).7 When soccer players suffer from mandible fractures, the subcondylar site is most frequently affected (28,6%).8
Horse riders, on the other hand, suffer most frequently from
fractures of the zygomatic bone (40%),9 while rugby players suffer
most frequently from mandible fractures (65%).10
Several authors stated that geographical differences might also
play a role in the frequency and type of sport-related maxillofacial
fractures.6,8,11 In Austria, 55.3% of sport-related mandible fractures
were caused by skiing,8 and in Switzerland 27% of sport-related
maxillofacial fractures were sustained during skiing and snowboarding.11 On the other hand, a study performed in the United
States reported no fractures due to skiing accidents.12 These geographical differences might be affected by different numbers of
individuals practicing specific sports.
Until now, no data are available on sport-related maxillofacial
fractures in the Netherlands. Therefore, the aim of the present study
was to explore the possible relation between the types of sport
practiced, and the frequency and nature of the facial bone fractures
of patients presenting in the oral-maxillofacial department of a
Dutch university center.
METHODS
This study is based on an analysis of a patient database from the
Department of Oral and Maxillofacial Surgery, Vrije Universiteit
University Medical Center (VUmc), Amsterdam, The Netherlands. The database consists of retrospectively collected data
from January 1, 2000 until January 1, 2010 and systematic
computer-assisted databases that have continuously recorded
patients with maxillofacial fractures between January 1, 2010
and April 1, 2014.
Both surgically and nonsurgically treated patients were included.
Only maxillofacial fractures caused by sports were included in this
study. The study was performed according to the guidelines of the
medical ethical committee of the Free University of Amsterdam.
Patients below the age of 4 and above the age of 80 were
excluded, as these patients were not expected to participate in
community sports. From the medical records, the following data
were retrieved: sex, age, type of sports, and type of maxillofacial
fracture. Maxillofacial fractures caused by bicycle accidents were
considered traffic accidents and were excluded from the study.
Fractures caused by skiing, snowboarding, and sled riding were
combined into winter sports. Baseball and softball fractures were
also combined into softball.
The maxillofacial fractures were divided into mandible fractures
(angle, body, condyle, guardsman fractures, coronoid process,
and symphysis), zygomatic complex fractures, mid-facial fractures
(Le Fort 1, 2, and 3, and alveolar process fractures of the maxilla),
orbital walls fractures (orbital and sphenoid sinus fractures), nasal
bone and frontal sinus fractures, skull fractures (parietal and
temporal bone fractures), and multi-trauma (2 or more trauma from
different complexes).
The IBM SPSS 21 package (IBM, Armonk, NY) was used to
analyze associations among multiple variables. Statistical significance was determined using x2, or Fisher exact test, if the sample
sizes were too small. P values <0.05 were considered statistically
significant.
RESULTS
The study population consisted of 108 patients with 128 maxillofacial fractures (79% male; 21% female). A mean age of 30.6 years
(SD, 12.0; range 1064) was observed. The highest incidence of
maxillofacial fractures was observed in subjects between 20 and
29 years old (Table 1).
#
TABLE 1. Age Distribution of Patients With Maxillofacial Fractures, Stratified According to Type of Sport Performed During the Accident
Soccer
Field
Hockey
Horse
Riding
3 (10%)
9 (30%)
10 (33%)
7 (23%)
1 (3%)
0 (0%)
30 (100%)
2 (7%)
17 (63%)
6 (22%)
1 (4%)
1 (4%)
0 (0%)
27 (100%)
5 (56%)
2 (22%)
1 (11%)
0 (0%)
1 (11%)
0 (0%)
9 (100%)
Age
1019
2029
3039
4049
5059
6069
Total
Rugby
Ice
Skating
Winter
Sports
Other
Sports
0 (0%)
6 (67%)
1 (11%)
2 (22%)
0 (0%)
0 (0%)
9 (100%)
1 (17%)
1 (17%)
3 (50%)
1 (17%)
0 (0%)
0 (0%)
6 (100%)
0 (0%)
1 (20%)
1 (20%)
1 (20%)
0 (0%)
2 (40%)
5 (100%)
2 (40%)
0 (0%)
2 (40%)
1 (20%)
0 (0%)
0 (0%)
5 (100%)
5 (29%)
3 (18%)
3 (18%)
4 (24%)
2 (12%)
0 (0%)
17 (100%)
TABLE 2. Frequency Distribution of Patients With Orofacial Fractures Stratified

According to Type of Sport Performed During the Accident
Type of Sport
Number of Patients (Percentage)
Soccer
Field hockey
Horse riding
Rugby
Martial arts
Ice skating
Cricket
Tennis
Bicycle racing
Winter sports, other than ice skating
Skateboarding
Inline skating
Ice hockey
Skydiving
Softball
Gymnastics
Go-karting
30
27
9
9
6
5
2
1
1
5
1
3
1
1
3
1
3
Count
(Percent Within
Age Intervals)
Martial
arts
(27.8%)
(25.0%)
(8.3%)
(8.3%)
(5.6%)
(4.6%)
(1.9%)
(0.9%)
(0.9%)
(4.6%)
(0.9%)
(2.8%)
(0.9%)
(0.9%)
(2.8%)
(0.9%)
(2.8%)
The patients had been engaged in 18 different sports as demonstrated in Table 2, where soccer has been the major cause of
maxillofacial trauma (28%) followed by field hockey (25%), horse
riding (8%), and rugby (8%). The most commonly sports observed
related maxillofacial fractures were zygomatic complex fractures
(45%), followed by mandible fractures (32%; Table 3). Further, no
significant differences were observed between the sport categories.
Soccer had the highest percentage of multitrauma (20%) followed
by field hockey (11%). Looking only at the mandible fractures, the
mandible body was mostly affected (45%), followed by mandible
condyle (36%; Table 4). Sports soccer and rugby were solely played
by males (Figure 1).
18
39
27
17
5
2
108
(17%)
(36%)
(25%)
(16%)
(5%)
(2%)
(100%)
DISCUSSION
This study confirms previous studies that sport is a major cause of
maxillofacial injuries. The most common sport-related fractures
were zygomatic complex fractures, followed by mandible fractures,
which is in accordance with results from previous studies.9 11,13,14
Nasal bone fractures were nearly absent in the present data, since
these fractures are usually treated by the ear nose throat (ENT)
department and therefore not included in the database used.
The highest incidence of sport-related maxillofacial fractures
occurred in individuals between the ages of 20 and 29 (Table 1).
Other studies found similar results, although the 36% in the present
study is slightly lower than the 41.4% to 52.9% in previous
studies.4,5,8,10,13,14 Most sport-related fractures occurred in males,
which is also in accordance with previous studies.7 10,13,14
Soccer and hockey were the most prominent causes of sportrelated maxillofacial trauma in the present study. This is in line with
the large number of people playing soccer in the Netherlands. Hockey,
on the other hand, is only the ninth most popular sport in the
Netherlands (Centraal Bureau Statistiek; CBS).15,16 However, field
hockey participation in the Amsterdam and the adjacent Amstelveen
suburb is high, with 6.5% of all Dutch hockey players playing in this
area (CBS). We suspect that this may contribute to the prominence of
hockey-related trauma in our data. However, this high number could
also be a result of hockey being a high-risk sport for maxillofacial
trauma. In Ireland,17 gaelic football was the sport responsible for most
fractures followed by cricket and soccer, respectively, while in
Japan10 and Great Britain18 rugby proved to be the main cause. In
Switzerland11 most fractures were sustained during skiing and snowboarding during team sports such as soccer or ice hockey and cycling.
In Brazil, nasal fractures were the most common soccer-related facial
fractures. In a retrospectively performed review about 451 Germans
soccer players who had suffered injuries during soccer games, the
head was affected in 23.9% of cases. The areas most frequently
involved were the facial and occipital regions.19
An interesting observation of the present study is that coronoid
process fractures were only observed in soccer players and not in
TABLE 3. Maxillofacial Fractures, Stratified According to Type of Sport Performed During the Accident
Fracture
Mandible
Zygoma complex
Midface
Orbital wall
Nasal bone / frontal sinus
Multiple
Total
Soccer
Field
Hockey
Horse
Riding
Rugby
Martial
arts
Ice
Skating
Winter Sports,
Other Than Ice Skating
Other
Sports
8 (27%)
14 (47%)
0 (0%)
1 (3%)
1 (3%)
6 (20%)
30 (100%)
10 (37%)
13 (48%)
0 (0%)
0 (0%)
1 (4%)
3 (11%)
27 (100%)
3 (33%)
4 (44%)
1 (11%)
0 (0%)
0 (0%)
1 (11%)
9 (100%)
2 (22%)
5 (56%)
0 (0%)
0 (0%)
2 (22%)
0 (0%)
9 (100%)
3 (50%)
1 (17%)
0 (0%)
1 (17%)
0 (0%)
1 (17%)
6 (100%)
0 (0%)
4 (80%)
0 (0%)
0 (0%)
0 (0%)
1 (20%)
5 (100%)
1 (20%)
2 (40%)
0 (0%)
2 (40%)
0 (0%)
0 (0%)
5 (100%)
7 (41%)
6 (35%)
0 (0%)
1 (6%)
0 (0%)
3 (18%)
17 (100%)
Total
34
49
1
5
4
15
108
(32%)
(45%)
(1%)
(5%)
(4%)
(14%)
(100%)
e93
TABLE 4. Location of Mandible Fractures, Stratified According to Type of Sport Performed During the Accident
Location
Angle
Body
Condyle
Coronoid process
Guardsman
Symphysis
Total
Soccer
Field
Hockey
Horse
Riding
Rugby
Martial
arts
Ice
Skating
0 (0%)
7 (47%)
5 (33%)
3 (20%)
0 (0%)
0 (0%)
15 (100%)
1 (8%)
8 (62%)
4 (31%)
0 (0%)
0 (0%)
0 (0%)
13 (100%)
0 (0%)
1 (25%)
2 (50%)
0 (0%)
1 (25%)
0 (0%)
4 (100%)
2 (67%)
1 (33%)
0 (0%)
0 (0%)
0 (0%)
0 (0%)
3 (100%)
1 (17%)
2 (33%)
2 (33%)
0 (0%)
0 (0%)
1 (17%)
6 (100%)
0
0
0
0
0
0
0
FIGURE 1. Some sports, such as soccer and rugby, were solely played by males.
other sports groups. This might be due to the fact that the most
common cause of accident in soccer is impact against another
player.10 However, in the study of Emshoff et al8 no fractures in the
coronoid region were observed among 28 fractures related to
soccer. Mandible angle fractures were more seen in rugby than
in other sports. Other studies also demonstrated that the mandible is
often a site of injury in rugby,9,10 but these previous studies did not
specify the frequency of mandible angle fractures in rugby players.
Other authors reported the most frequent fracture site of the
mandible was the angle followed by the symphysis in maxillofacial
fractures sustained during sports played with ball.20
The present study has several potential limitations. In the first
place, it is a single-center study. Amsterdam has 3 other hospitals
where patients with maxillofacial injuries are treated. As the
patients are not equally divided into the 4 hospitals in Amsterdam,
some hospitals may see more and different kinds of patients than the
other hospitals. Therefore, the results in the present study might not
be fully representative for the Netherlands. As the data were partly
collected retrospectively, this may also have introduced information
bias. Nevertheless, the results found in this study are mostly in line
with other studies, which suggest that the data might be useful for
the development of protocols to prevent maxillofacial trauma in
certain sports.2
In conclusion, the results of this study suggest a relation between
type of sport and the nature and frequency of the fractures it causes.
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fractures. J Craniofac Surg 2011;22:16311634
2. Van den Bergh B, Karagozoglu KH, Heymans MW, et al. Aetiology and
incidence of maxillofacial trauma in Amsterdam: a retrospective
analysis of 579 patients. J Craniomaxillofac Surg 2012;40:165169
3. Salentijn EG, Van den Bergh B, Forouzanfar T. A ten-year analysis of
midfacial fractures. J Craniomaxillofac Surg 2013;41:630636
4. Gassner R, Bosh R, Tuli T, et al. Prevalence of dental trauma in 6000
patients with facial injuries: implication for prevention. Oral Surg Oral
Med Oral Pathol Radiol Endod 1999;87:2733
e94
(0%)
(0%)
(0%)
(0%)
(0%)
(0%)
(0%)
Winter Sports,
Other Than Ice Skating
Other
Sports
Total
0 (0%)
1 (100%)
0 (0%)
0 (0%)
0 (0%)
0 (0%)
1 (100%)
1 (8%)
5 (38%)
7 (54%)
0 (0%)
0 (0%)
0 (0%)
13 (100%)
5 (9%)
25 (45%)
20 (36%)
3 (5%)
1 (2%)
1 (2%)
39 (100%)
5. Elhammali N, Bremerich A, Rustemeyer J. Demographical and clinical

aspects of sports-related maxillofacial and skull base fractures in
hospitalised patients. Int J Oral Maxillofac Surg 2010;39:857862
6. Kunamoto DP, Maeda Y. A literature review of sports-related
maxillofacial trauma. Gen Dent 2004;52:270280
7. Goldenberg DC, Dini GM, Pereira MD, et al. Soccer-related facial
trauma: multicenter experience in 2 Brazilian university hospitals. Plast
Reconstr Surg Glob Open 2014;2:e168
8. Emshoff R, Schoning H, Rothler G, et al. Trends in the incidence and
cause of sport-related mandibular fractures: a retrospective analysis.
J Oral Maxillofac Surg 1997;55:585592
9. Frenguelli A, Ruscito P, Bicciolo G, et al. Head and neck trauma in sporting
activities. Review of 208 cases. J Craniomaxillofac Surg 1991;19:178181
10. Tanaka N, Hayashi S, Amagasa T, et al. Maxillofacial fractures
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11. Exadaktylos AK, Eggensperger NM, Eggali S, et al. Sports related
maxillofacial injuries: the first maxillofacial trauma database in
Switzerland. Br J Sports Med 2004;38:750753
12. Soporowski NJ, Tesini DA, Weiss AI. Survey of maxillofacial sportsrelated injuries. J Mass Dent Soc 1994;43:1620
13. Cerulli G, Carboni A, Mercurio A, et al. Soccer-related
craniomaxillofacial injuries. J Craniofac Surg 2002;13:627630
14. Mourouzis C, Koumoura F. Sports-related maxillofacial fractures: a
retrospective study of 125 patients. Int J Oral Maxillofac Surg
2005;34:635638
15. Centraal Bureau Statistiek.Available at: http://www.cbs.nl. Accessed
June 25, 2014
16. Koninklijke Nederlandse Hockey Bond,Available at: http://www.knhb.nl.
Accessed June 25, 2014
17. Fasola AO, Obiechina AE, Arotiba JT. Sports-related maxillofacial
fractures in 77 Nigerian patients. Afr J Med Med Sci 2000;29:215217
18. Hill CM, Burford K, Martin A, et al. A one-year review of maxillofacial
sports injuries treated at an accident and emergency department. Br J
Oral Maxillofac Surg 1998;36:4447
19. Kolodziej MA, Koblitz S, Nimsky C, et al. Mechanisms and
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sustained during sports played with a ball. Oral Surg Oral Med Oral
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Le Fort II Setback Osteotomy to

Correct Naso-Ethmoido-Maxillary
Protrusion
Sandra Konopnicki, MD, MSc, Romain Nicot, MD, MSc,
Gwenael Raoul, MD, PhD, and Joel Ferri, MD, PhD
Background: Marked class II dentofacial deformity associated
with centrofacial protrusion may be difficult to treat successfully.
#
The purpose of this article was to report on Le Fort II setback

osteotomy (LIISBO) to correct Naso-Ethmoido-Maxillary Protrusion (NEMP), to describe its indications and surgical techniques,
and to analyze aesthetic and occlusal changes.
Materials and methods: From November 2011 to November
2014, patients with NEMP, treated with LIISBO, were included
in the study. Cephalometric analysis of Delaire was performed
before and 1 year after surgery. Skeletal and soft tissues movements
were measured between preoperative and postoperative lateral
cephalographs.
Results: Fourteen patients were treated in our department by
LIISBO. Ten patients were analyzed and presented a stable class
I occlusion with reliable aesthetic results. The mean maxillary
setback was 2.8 mm at nasopalatal point (Np), 3.1 mm at A
point, and 3.7 mm at Pti (inferior pterygomaxilar point). The mean
maxillary impaction was 2.4 mm at Np, 3 mm at A point, and
0.6 mm at Pti. The B, mental, and pogonion points showed
an advancement with an average of 7.4, 7.9, and 7.7 mm,
respectively. Measured soft tissues variations showed a backward
movement of the nasal tip, the subnasal point, and the upper lip of
1.5, 1.6, and 0.7 mm, respectively. The lower lip, sublabial
point, and the skin pogonion were advanced by 3.2, 5.4, and
6.2 mm, respectively.
Conclusions: Le Fort II setback osteotomy may be regarded as the
ideal treatment for adult patient presenting a NEMP syndrome.
Key Words: Class II malocclusion, Le Fort II osteotomy,
maxillary protrusion, maxillary setback, nasal protrusion,
orthognatic surgery
owadays, orthodontico surgical treatment for dentofacial

dysmorphosis has well-defined indications in most cases,
but the management of some patients remains problematic.
Le Fort I and Le Fort II maxillary advancement can be used in
the surgical treatment of facial dysmorphosis. Le Fort II osteotomy
was first reported by Converse in 1970,1 and then technical refinements were proposed by Kufner and Henderson and Jackson2,3,4 in
cases of nasomaxillary retrusion or hypoplasia.
Classic management of class II malocclusion with skeletal
deformity is bilateral sagittal splinting of the mandible, associated
in some cases with maxillary osteotomy, to treat vertical and/or
transversal discrepancies of the maxilla. This treatment, however,
can have an incomplete outcome for the face morphology and
occlusal stability in case of marked maxillary protrusion. This
situation leads some authors to propose a Le Fort I maxillary push
back movement in particular class II.5 Although this particular
osteotomy has its specific indications, some cases in which the
whole ethmoido-naso-maxillary complex is protrused must be
treated differently because of the adverse aesthetic consequences
From the Department of Oral and Maxillofacial Surgery, Roger Salengro
Hospital, Lille 2 University, Lille, France.
Received February 8, 2015.
Address correspondence and reprint requests to Sandra Konopnicki,
Service de Chirurgie Maxillofaciale et Stomatologie, Hopital Salengro, Centre Hospitalier Regional Universitaire de Lille, rue Emile
laine, 59000 Lille, France; E-mail: sandra.konopnicki@gmail.com
ISSN: 1049-2275
DOI: 10.1097/SCS.0000000000002243
#
that a Le Fort I push back osteotomy would induce, such as

increasing the nasal protrusion.6 Therefore, we think that the Le
Fort II maxillary setback osteotomy (LIISBO) can play a role in the
treatment of these cases.
The purpose of this article was to report a new bimaxillary
osteotomy including LIISBO to correct the Naso-EthmoidoMaxillary Protrusion (NEMP), to describe its indications and
surgical techniques, and to analyze the resulting aesthetic and
occlusal changes.

Patients
From November 2011 to November 2014, patients with NEMP,
treated with bimaxillary osteotomy including LIISBO, were
included in the study. Patients were informed about the LIISBO
associated with mandibular advancement surgical treatment, and
the informed consent was obtained from all the patients.
Treatment Plan
Preoperative evaluation of the patients was clinical and radiologic. Examinations were performed at the beginning of the
orthodontic treatment and 1 month before surgery. Models and
systematic pictures of the patients were realized. Orthopantogram,
lateral, and facial cephalograms were performed and analyzed using
architectural and structural analysis of Delaire.7,8 Indications were
based on clinical and radiologic criterions:
Clinical evaluation: Marked skeletal class II deformity with
convex profile and nasal protrusion. Class II malocclusion molar
and canine, and exoclusion in some cases because of transversal
excess of the maxilla.
Radiologic evaluation: Delaires architectural and structural
cephalometric analysis showed a NEMP associated with mandibular hypoplasia in some cases. As described previously,9 the NEMP
is characterized by a class II skeletal deformity with convex profile,
a sagittal hyperplasia of the maxilla associated with centrofacial
protrusion including nasal protrusion. Maxillary hyperplasia may
be more pronounced in the premaxilla associated with an upper
alveolar prognathism. Transversal and vertical excess of the maxilla
may be associated. The craniofacial base line, drawn from the M
point (junction of the frontonasal, maxillofrontal, and maxillonasal
sutures) through the Cp point (condyle posterior), is always
increased. In addition, an inward inclination of the upper incisors,
an outward inclination of the lower incisors, and a lower alveolar
retrusion may be observed.
Surgical Procedure
In all the cases, orthodontical treatment was used to prepare
patients occlusion to the intervention and a single surgeon achieved
all the procedures. All procedures were realized under general
anesthesia with controlled hypotension and nasotracheal intubation.
The incision of the mucosa in the vestibular fold was from 15 to 25
after a subperiostal infiltration with adrenalined saline solution
(average amount 10 mL). The entire anterior surface of the maxilla
was exposed by subperiosteal dissection and a full-thickness flap
elevated superiorly; a special care was taken of the infraorbital
nerve. Then, the maxillar tuberosity and piriform aperture were
exposed. The nasal fossa floor mucoperiosteum was elevated; the
fibrous connection of the cartilaginous septum and the osseous
connection of the vomer were separated from both maxilla and
palatine bone. The maxillary rostrum was then removed with a
chisel. As for the Le Fort I procedure, the osteotomy was performed
bilaterally, with oscillating saw from maxilla tuberosity to below the
e95
FIGURE 1. Surgical procedure: A, monobloc osteotomy (one part); B, outline

bone cuts using 2 parts osteotomy.
FIGURE 3. Lateral cephalometric radiograph of patient 7 before (left) and after

surgery (right) analyzed using architectural cephalometric analysis of Delaire.
FIGURE 4. Lateral cephalometric radiograph: x and y axes measured skeletal

and cutaneous point.
FIGURE 2. Backward translation of the maxilla after cutting the pterygoid

processes.
osteotomies: first, a complete Le Fort I osteotomy was performed.

Then, the vertical osteotomy of the centrofacial area was performed
from the Le Fort I section to the frontonasal suture (Fig. 1B).
Depending on the case some patients received other additional
procedures such as segmental osteotomies and genioplasties
(Table 1).
emergence of the infraorbital nerve. From this point, the cut was
completed vertically with a bur to the frontal process of the maxilla.
Then, a transverse section was made across the frontonasal suture
(Fig. 1). The procedure was performed bilaterally. This osteotomy
made with the oscillating saw, and the bur was completed with
chisel. Separations of the maxilla from the pterygoids were accomplished with a broad curved chisel. The osteotomy line was opened
widely with expansion forceps into the maxilla-zygomatic buttress.
Maxilla was pulled down using Rowes maxillary disimpaction
device. The difficulty was the access to the maxilla posterior
structures. The descending palatine vessels were dissected to access
the pterygoid processes. The lower ends of the pterygoid processes
were then cut. Maxilla was then pushed backward according to the
planed translation (Fig. 2). The bony fixation was made by L-plates
(Modus, Medartis, Basel, Switzerland) with 4 screws on canine
pillars and 2 screws on maxillo-zygomatic pillars. In addition, a
miniplate was fixed to the frontal process of the maxilla.
The bilateral sagittal mandibular split was made according to
Epkers osteotomy technique and the mandibular advancement realized. Maxillo-mandibular elastic fixation was maintained for 10 days.
Analyses
The postoperative clinical control consisted in a visit at day 10, 1
month, 6 months, and 1 year postoperative and pictures were taken.
Radiologic examination including cephalograms and orthopantograms was performed 1 year after surgery and analyzed using the
architectural and structural analysis of Delaire6,7 (Fig. 3). Skeletal
and soft tissues movements were measured comparing preoperative
and postoperative lateral cephalographs. The cranial line C1 was
used for x-axis. C1 line is the superior cranial base line drawn from
the M point (junction of the frontonasal, maxillofrontal, and maxillonasal sutures) through the clinoid point (Cl). The y-axis was
extended from the clinoid point downward. Backward and upward
movements were reported with negative values (Fig. 4).
Bony movements were characterized using the following point
(Fig. 4): Is: incision superius (midpoint on the incisal edge of the most
proeminent upper incisor), Ii: incision inferius (midpoint on the incisal
edge of the most prominent lower incisor), A: subspinal (deepest point
of the anterior contour of the upper alveolar arch); B: supramentale
(deepest point on the anterior contour of the lower alveolar arch), Np:
superior foramen of the nasopalatal canal; Pti PNS: pterygomaxillar posterior nasal spine (dorsal surface of the maxilla, at the level
Technical Refinements
1. First patient received a Z cutaneous incision on the glabella,
providing a good access to the nasal bridge. This incision was not
necessary for other cases.
2. First 2 patients underwent monobloc Le Fort II osteotomy as
described above (Fig. 1A). All other patients received 2 segments
TABLE 1. Patient and Surgery Characteristics, Lateral Cephalometric Analysis of Delaire Before and After Surgery
Before Surgery
Patients
1
2
3
4
5
6
7
8
9
10
After Surgery
Surgery
Sex
Age
C1/F1
C1/F1M
C1/F1m
C1/F1M
C1/F1m
Monobloc, BSSO
Monobloc, AI, BSSO
2 parts, BSSO, genioplasty, SO lower incisor
2 parts, BSSO, AI
2 parts, TSMO, SO upper incisor
2 parts, BSSO, AI
2 parts, BSSO, SO lower incisor
2 parts, BSSO, genioplasty
2 parts, BSSO, AI
2 parts, BSSO
M
F
M
F
M
F
M
F
M
M
35
30
16
37
39
25
17
21
17
18
92
85
89
85
91
87
90
85
89
89.5
97
93
92
93
93
87
95
89
89
95
93
84
84
84
90
83
87
76
84
85
92
89
90
91
90
87
90
85
89
90
92
86
90
91
90
87
90
85
89
88
AI, anterior impaction; BSSO, bilateral sagittal split osteotomy; F, female; F1M, facial maxillary line from FM to the Np point; F1m: facial mandibular line from FM to Me point;
M, male; SO, segmental osteotomy; TSMO, total subapical mandibular osteotomy C1/F1: craniofacial balance line.
e96
FIGURE 5. Photographs of patient 1 who underwent a monobloc osteotomy

before (left) and after (right) surgery.
of the nasal floor, anterior limit of the pterygo-palatine fossa), Me:

mental (intersection of the anterior part of the mandibular inferior
border and the posterior line of the mandibular symphysis), Pog:
pogonion (most prominent point of the chin).
The following points were used to assess the soft tissues movements (Fig. 4): Pn: the anterior point of the nasal tip, Sn: the
subnasal point, Ls; labial superior (the anterior point of the upper
lip), Li: labial inferior (the anterior point of the lower lip), Sl:
sublabial, pog: skin pogonion (the anterior point of the chin).
RESULTS
Fourteen patients (5 women and 9 men) were treated in our
department by LIISBO with additional mandibular advancement.
One-year follow-up was available for 10 patients to analyze bony
and soft tissues movements. Six months and 1 year after surgery, all
patients presented a stable class I occlusion. Table 1 shows patient
characteristics, surgical procedure, and lateral cephalometric
analysis, before and after surgery. Before surgery, 8 patients had
a maxillary protrusion. Two patients (patients 6 and 9) did not show
a maxillary protrusion C1/F1M C1/F1, but presented a premaxillary protrusion with an increase length of the line from anterior
nasal spine to nasopalatal point (Np) and an upper alveolar prognathism. All patients presented a mandibular retrusion except one
showing a bimaxillary protrusion but higher for the maxilla (patient
#
FIGURE 6. Pictures of patient 7 before (left) and after (right) 2 segments surgery.
1). One year after surgery, all patients presented a class I occlusion.
Of the 10 patients, 8 showed a skeletal class I. The 2 remaining
patients presented a short retrusion of the Me point after surgery,
resulting on a skeletal class II, because of an outward inclination of
the lower incisors without orthodontic or surgical correction, which
has limited the bony movement (patients 2 and 10). Moreover, a
genioplasty was not performed for those 2 patients. Two other
patients presented an outward inclination of the lower incisors,
which was corrected with a segmental osteotomy (patients 3 and 7).
One patient had a segmental osteotomy of the upper incisors to
correct an inward inclination and a total subapical mandibular
osteotomy because of an inferior alveolar retrusion associated
(patient 5). Two patients had a genioplasty. Photographs of patients
who underwent monobloc and 2 segment osteotomies are shown,
respectively, in Figures 5 and 6.
Table 2 shows the skeletal and soft tissues movements. The
mean maxillary setback was 2.8 mm at Np, 3.1 mm at A point,
and 3.7 mm at Pti, whereas the mean backward movement of the
incisal edge of the upper incisor was 0.2 mm. The mean maxillary
impaction was 2.4 mm at Np, 3 mm at A point, and 0.6 mm at
Pti, whereas 2.9 mm upward movement of the upper incisor was
measured. The upward translation was higher at the anterior part of
the maxilla because of an anterior impaction in 4 cases. The first
patient showed a downward movement of the maxilla because of a
monobloc anterior clockwise rotation of the maxilla. Regarding
e97
TABLE 2. Bone and Soft Tissues Movements

Upper Jaw
Skeletal Points
Mean
x-axis
y-axis
Cutaneous points
Mean
x-axis
y-axis
Lower Jaw
Is
Np
Pti
Ii
Me
Pog
0.2
2.9
3.1
3
2.8
2.4
3.7
0.6
5.5
1
7.4
1.5
7.9
7
7.7
5
Ls
Sn
Pn
Li
Sl
Pog
0.7
1.1
1.6
0.2
1.5
1.2
3.2
1.9
5.4
1.8
6.2
0.5
A, subspinal; B, supramentale; Ii, incision inferius, incisal edge of the most proeminent lower incisor; Is, incision superius, incisal edge of the most proeminent upper incisor; Li,
labial inferior (lower lip); Ls, labial superior (upper lip); Me, mental; Np, nasopalatal canal; Pti PNS: pterygomaxillare posterior nasal spine; Pn, nasal tip; Pog, pogonion; Pog,
skin pogognon (anterior point of the chin); Sl, sublabial; Sn, subnasal. Mean postoperative-preoperative variations (mm).
mandibular advancement, all patients presented an anterior translation of the B point with a mean of 7.4 mm. The Me and pog point
showed an advancement with an average of 7.9 and 7.7 mm,
respectively. One patient (patient 5) did not show any modifications
of the Me and Pog while 3 mm advancement of the Ii and B point
because of a total subapical mandibular osteotomy.
Measured soft tissues variations showed a backward movement of
the nasal tip, the subnasal point, and the upper lip of 1.5, 1.6, and
0.7 mm, respectively. The lower lip, sublabial point, and the skin
pogonion were advanced by 3.2, 5.4, and 6.2 mm, respectively.
One patient presented a nasolacrimal duct dysfunction.
Mandibular screws placed at the inferior segmental osteotomy were
removed for 1 patient because of an infection. The second patient
showed a nasal deviation.
DISCUSSION
As we demonstrated, LIISBO appears to be of interest in precise cases
of facial dysmorphosis. Functional deformities such as anterior
rotation of the maxilla or posterior rotation of mandibular ramus
can be successfully treated with precocious orthopedic care during
childhood.10 Constitutional anomalies, such as excessive length of
the premaxilla or maxillary vertical dimension excess, should frequently be treated in a combined orthodontic-surgical approach;
orthopedic care alone having poor outcomes in those cases.11
Classically, dentofacial class II deformity was usually treated by
orthodontic treatment or combined surgical and orthodontic
sequences, consisting in extraction of maxillary premolars with
anterior maxillary osteotomy.12,13 Suitable occlusal results were
observed but unaesthetic, resulting in facial disharmony.12,13 Nowadays in maxillary skeletal protrusion, bilateral sagittal split osteotomy and mandibular advancement either with or without maxillary
osteotomy are the most usual treatment. The maxillary osteotomy is
performed depending on the vertical and transversal discrepancies.
Le Fort I maxillary setback osteotomy has been described,
combined with sagittal mandibular advancement for correction
of class II with marked maxillary protrusion. Colantino and
Dudley14 first described the total maxillary setback osteotomy in
1970 (5 mm setback) and Cruickshank in 197215 (14 mm setback).
Le Fort I setback osteotomy is useful for patients with marked
maxillary prognathism, anterior vertical excess, acute nasolabial
angle, and gummy smile.5,6 Chouet-Girard and Mercier6 reported a
backward translation of the upper lip, a mean opening of the
nasolabial angle after maxillary setback without movements of
the nasal tip. Therefore, Le Fort I setback osteotomy increases
the nasal protrusion and should be contraindicated for patients with
centrofacial protrusion and obtuse nasolabial angle.6 Some specific
e98
maxillary protrusions are particular. They have been described as

NEMP, a specific malocclusion.9 This dysmorphosis is related to a
constitutional overgrowth of the central part of the face, which is at
the origin of a global protrusion of the ethmoid, the nose, and the
maxilla. In typical cases, the overgrowth affects sagittal and
tranversal dimensions.9 For this specific malocclusion, during
childhood, premolar extractions must be avoided to prevent poor
aesthetic outcomes. At the adult age maxillary setback provides
stable occlusion, but leads to unaesthetic outcomes because of an
excessive protrusion aspect of the nose that the setback movement
of the maxilla had increased. Therefore, when those major discrepancies are associated with a centrofacial protrusion, in some
limit cases, LIISBO was proposed to our patients.
Le Fort II advancement osteotomy was described for maxillary
hypoplasia either with or without malar hypoplasia. First description of the anterior Le Fort II osteotomy was reported by Converse
in 1970.1 In 1973, Henderson and Jackson3 described the pyramidal
Le Fort II osteotomy following the lines of Le Fort II fracture. In
those techniques, the osteotomy line crosses the inferior and lateral
orbital rims, and leads to complications such as diplopia, or
lacrymal duct dysfunction.4 Kufner reported a quadrangular Le
Fort II osteotomy in 1971 to treat associated malar hypoplasia.2
This report is the first using LIISBO. In our technique, we did
not use an orbital approach. The osteotomy did not touch the orbital
frame, preventing diplopia and nasolacrimal duct dysfunction,
encountered in previous Le Fort II osteotomy techniques. One
patient, however, underwent a nasolacrimal duct dysfunction,
probably because of the placement of the screw.
The limits of this surgical LIISBO procedure consist on the
difficulty of the technique. The access to the pterygoids may be
complex. In one of the 2 cases of monobloc osteotomies, we
observed a nasal deviation. That is explained by a discrepancy
between the nose and the teeth leading to the impossibility of having
a central positioning of the teeth and the nose with a one-piece
monobloc osteotomy. Moreover, the movement observed in the first
patient was more likely a clockwise rotation than a backward
translation. That can be explained by the difficulty we faced to
mobilize the osteotomized maxilla in all directions. Indeed, it is
sometimes difficult to do an impaction and a setback displacement
with a one-piece Le Fort II ostotomy. Therefore, we avoided those
difficulties by using a 2 parts osteotomy. This technique was more
repeatable, gave the opportunity to perform a vertical control movement such as anterior impaction, and to realize different setback of the
maxilla and the nose. For 1 patient, an incision was made in the
glabella to allow the access of the frontonasal suture (patient 1).
Although we obtained satisfying healing with a discreet scar (Fig. 5),
it could result in poor aesthetic outcomes for the patients. Coronal
#
incision or bilaterally paralateronasal incision described for Le Fort II

advancement is in our opinion too invasive for that functional
orthognatic surgery. Therefore, for the other patients, we used only
an intraoral approach. This approach can combine both satisfactory
access to osteotomy sites and deceit of the scar. Other approaches
described, such as nasal degloving, are unnecessary.16,17
As described in the Le Fort I setback,5 a stable class I occlusion
with no relapse was observed at 1 to 4 years follow-up. The mean
setback of the subnasal point was almost equal to the nasal tip
movement and the upper lip moved only at an average of 0.7 mm.
Consequently, the nasal protrusion does not increase after the
procedure and may be attenuated, showing excellent aesthetic
outcomes. The shorter movement of the upper lip is correlated
to the outward translation of the upper incisor when segmental
osteotomy or anterior impaction was performed.
The NEMP is a constitutional anomaly because of an excessive
growth of the chondrocranium and the septo-ethmoid.9 Before the
nasal growth, it may be observed showing a marked convex profile
and an increased length of the anterior basal cranial line. The septoethmoid inordinate growth result in centrofacial protrusion, associated with a protrusion of the premaxilla.10,18,19,20 Therefore, an
alveolar protrusion with inward inclination of the upper incisor may
be observed associated in some cases with an inferior alveolar
retrusion. For some patients, incisor abnormal inclination could not
be corrected by orthodontic treatment. Consequently, segmental
osteotomies were performed to correct the dental inclination. One
patient (patient 5) presented an inferior alveolar retrusion associated
with a subnormal mandibular position corrected by using a subapical osteotomy technique.
To conclude, the LIISBO, or the Le Fort I with nasomaxillary
setback, can be considered as the ideal treatment for adult patient
presenting a NEMP. Further study comparing LIISBO to standard
osteotomy should be performed to show the superior occlusal
stability and aesthetic outcomes.
REFERENCES
1. Converse JM, Horowitz SL, Valauri AJ, et al. The treatment of
nasomaxillary hypoplasia. A new pyramidal naso-orbital maxillary
osteotomy. Plast Reconstr Surg 1970;45:527535
2. Kufner J. Four-year experience with major maxillary osteotomy for
retrusion. J Oral Surg 1971;29:549553
3. Henderson D, Jackson IT. NASO-maxillary hypoplasisthe Le Fort II
osteotomy. Br J Oral Surg 1973;11:7793
4. Steinhauser EW. Variations of Le Fort II osteotomies for correction of
midfacial deformities. J Maxillofac Surg 1980;8:258265
5. Schouman T, Baralle MM, Ferri J. Facial morphology changes after
total maxillary setback osteotomy. J Oral Maxillofac Surg 2010;68:
15041511
6. Chouet-Girard F, Mercier J. [Total osteotomy for maxillary setback.
Indications, technique, results]. Rev Stomatol Chir Maxillofac
2003;104:317325
7. Delaire J. [Architectural and structural craniofacial analysis (lateral
view). Theoretical principles. Some example of its use in maxillofacial
surgery (authors transl)]. Rev Stomatol Chir Maxillofac 1978;79:133
8. Delaire J, Schendel SA, Tulasne JF. An architectural and structural
craniofacial analysis: a new lateral cephalometric analysis. Oral Surg
Oral Med Oral Pathol 1981;52:226238
9. Konopnicki S, Nicot R, Sauve C, et al. [Naso-ethmoido-maxillary
protrusion (NEMP): a specific dysmorphosis]. Rev Stomatol Chir
Maxillofac Chir Orale 2014;115:9499
10. Delaire J. [Considerations on facial growth (particularly of the maxilla).
Therapeutic deductions]. Rev Stomatol Chir Maxillofac 1971;72:5776
11. Schendel SA, Eisenfeld JH, Bell WH, et al. Superior repositioning of
the maxilla: stability and soft tissue osseous relations. Am J Orthod
1976;70:663674
#
12. Seward GR. The treatment of class II facial deformity. Maxillary

operations. Br J Oral Surg 1973;10:254264
13. Proffit WR, White RP. Combined orthodontic and surgical management of
maxillary protrusion in adults. Am J Orthod 1973;64:368383
14. Colantino RA, Dudley T. Correction of maxillary prognathism by
complete alveolar osteotomy. J Oral Surg 1970;28:543548
15. Cruickshank GW, Pankow CW, Colarusso DA. A total maxillary
osteotomy and retropositioning of the maxilla: report of a case.
J Oral Surg 1972;30:586588
16. Kinnebrew MC, Zide MF, Kent JN, et al. Fort II Procedure for
simultaneous correction of maxillary and nasal deformities. J Oral
17. Wedgewood D. An approach to Le Fort II osteotomy. Br J Oral
18. Couly G. [The human cartilaginous mesethmoid (authors transl)]. Rev
Stomatol Chir Maxillofac 1980;81:135151
19. Salagnac JM. [Division-2 class 2. The identification and classification
of different clinical variations using J Delaires craniofacial
architectural and structural analysisthe therapeutic deductions].
Orthod Fr 1982;53:623640
20. Paranque AR. [The craniofacial architectural factors predisposing to a
skeletal Class II identified by Jean Delaires architectural analysis]. Rev
Stomatol Chir Maxillofac 2000;101:311
Use of a Titanium Microplate to

Anchor Subunit Reconstruction
at the NasalCheek Junction
Marcin Czerwinski, MD, PhD and Edward M. Gronet, MD
Abstract: Reconstruction of combined nose, cheek, and/or
inferior eyelid defects is facilitated by stable anchorage at
the nasal cheek junction. The previously reported techniques
of drill holes and Mitek anchors are not without disadvantages.
The authors present a simple means of anchoring soft tissue
flaps at the nasal cheek junction: a titanium miniplate secured
with a screw at each end. Our case report describes successful,
lasting, and complication-free anchorage of cheek, forehead,
and eyelid flaps to a single miniplate placed along the piriform
aperture.
Key Words: Nasal reconstruction, cheek reconstruction,
naso-facial junction
ombined defects of the nose, cheek, and/or inferior eyelid

are commonly encountered following facial cancer ablation
or traumatic injuries. Optimal aesthetic outcome requires independent reconstruction of each topographic unit. Their connection at the nasalcheek junction should restore its natural
From the Baylor Scott & White, Division of Plastic Surgery, Department of
Surgery, Temple, TX.
Address correspondence and reprint requests to Marcin Czerwinski, MD,
Baylor Scott & White, Division of Plastic Surgery, Department of
Surgery, 2401 S. 31st Street, Temple, TX 76508;
E-mail: mczerwinski@sw.org
ISSN: 1049-2275
DOI: 10.1097/SCS.0000000000002244
e99
concavity while preventing lateral displacement of the nasal ala

and infero-lateral displacement of the lower eyelid by the pull of
the cheek reconstructive flap.1 To achieve these goals reliably,
stable anchorage of the reconstructive flaps to the underlying
skeleton is suggested. Previously reported techniques of nasal
cheek junction skeletal anchorage include placement of multiple
drill holes in the maxilla and the use of Mitek anchors.2,3
Although both methods are effective, they have significant
disadvantages: drill holes make passage of needles without tearing
nasal mucosa difficult, multiple Mitek anchors may fracture the thin
piriform rim, may migrate intranasally, and are expensive ($450
each). We present a different approach to anchoring flaps at the
nasalcheek junction by using a titanium microplate (Synthes,
West Chester, PA). We believe this technique is advantageous
because of its speed, technical simplicity, and provision of multiple
fixation sites with minimal risk of underlying osseous fracture or
intranasal migration. The cost of the microplate and 2 screws is
$850, which is cheaper than the minimum of 2 Mitek anchors that
have to be used.
FIGURE 2. From left to right: 110 10 cm right upper lip, right cheek, and
right nasal side wall defect in a 49-year-old female following Mohs excision of a
basal cell carcinoma; 2right cheek and left paramedian forehead flaps in
position anchored to a 6 hole 0.4 mm titanium miniplate (Synthes, West
Chester, PA) at the piriform rim, demonstrating sharpness of the naso-facial
junction; 3 and 43-month postoperative frontal and oblique photographs
demonstrating continued sharpness of the naso-facial junction and lack of
alar distortion.
METHODS
We retrospectively identified 4 patients who underwent reconstruction of a combined nose, cheek, and/or inferior eyelid defect
with anchorage at the nasalcheek junction using a titanium
microplate (Synthes). This report seeks to illustrate the surgical
technique used.
SURGICAL TECHNIQUE
Standard flaps for reconstruction of the nose, cheek, and/or inferior
eyelid are chosen and elevated. If any soft tissue at the piriform rim
is present, it is elevated in a subperiosteal plane, exposing an area
for plate placement. A 4 to 8 hole 0.4 mm titanium microplate is
selected and placed at and parallel to the piriform rim with 2 of the
4 mm screws, the first above and medial to the maxillary canine root
and the second inferior to the medial canthus. The plate is not placed
flush with the bone, but instead a small amount of space is left
underneath in the center to facilitate needle passage. All sutures
from each flap to the plate are passed according to the optimal
vector necessary before being tied.
CLINICAL PRESENTATIONS
Four representative cases with long-term results are shown in
Figures 14.
DISCUSSION
Combined defects of the nose, cheek, and/or inferior eyelid are
challenging to restore aesthetically. Independent reconstruction of
FIGURE 1. From left to right: 1right cheek and nasal ala defect in a 73-yearold female following Mohs excision of a basal cell carcinoma; 2right cheek
and right paramedian forehead flaps in position anchored to a 4 hole 0.4 mm
titanium miniplate (Synthes, West Chester, PA) at the piriform rim,
demonstrating sharpness of the naso-facial junction; 3 and 43-month
postoperative frontal and oblique photographs demonstrating continued
sharpness of the naso-facial junction and lack of alar distortion.
e100
FIGURE 3. From left to right: 1nasal tip, dorsum, left nasal side wall, left ala,
left cheek defect in a 46-year-old female following basal cell carcinoma excision;
2left cheek, left paramedian forehead flaps, septal door flaps in position
anchored to a 6 hole 0.4 mm titanium miniplate (Synthes, West Chester, PA) at
the piriform rim, demonstrating sharpness of the naso-facial junction; 3 and
4immediate postoperative frontal and oblique photographs demonstrating
continued sharpness of the naso-facial junction and lack of alar distortion.
each facial unit with their stable anchorage at the nasalcheek

junction appears to provide optimal outcomes. Anchorage helps
prevent blunting of the nasalcheek junction, asymmetric alar
widening, and lower eyelid malposition with potential for subsequent lagophthalmos and lacrimal system dysfunction.
Different methods for nasalcheek junction skeletal anchorage
have been described, including the use of multiple drill holes and
Mitek anchors at the level of the piriform rim.2,3 Drill holes do not
allow for easy needle passage without tearing of nasal lining
because of the acute angulation of the piriform rim. Mitek anchors
end up on the medial aspect of the piriform rim because of its
thinness, with the potential for eventual erosion through the nasal
lining. With either method, risks of piriform rim fracture, technical
awkwardness, and costs rise as the number of anchoring points
increases. A titanium microplate placed at the piriform rim allows
for the placement of an unlimited number of sutures with technical
FIGURE 4. From left to right: 16 5 cm right nasal sidewall, right cheek, and
inferior eyelid defect in a 73-year-old male following Mohs excision of a basal
cell carcinoma; 2right cheek and right paramedian forehead flaps in position
anchored to a 5 hole 0.4 mm titanium miniplate (Synthes, West Chester, PA)
at the piriform rim, demonstrating sharpness of the naso-facial junction; 3 and
43-month postoperative frontal and oblique photographs demonstrating
continued sharpness of the naso-facial junction, lack of alar, and inferior eyelid
distortion.
#
ease and speed, allowing for selection of optimal vectors for an

aesthetic reconstruction. Furthermore, as the points of skeletal
fixation are in the thick alveolar bone and frontal process of the
maxilla, the risk of osseous fracture is minimal.
Although we have not noted any negative sequelae of this
technique in our experience, we do acknowledge that the procedure
is not without risks. First, the insertion of a foreign body increases the
risks of infection and exposure with the potential for future removal.
Second, if adjuvant radiation therapy is required, the plate may
theoretically reduce the effective dose immediately underneath it.
REFERENCES
1. Honda K, Reichel J, Odland P. Anchored rotation flap for infraorbital
cheek reconstruction: a case series. Dermatol Surg 2007;33:516520
2. Mathijssen IM, Roche NA, Vaandrager JM. Soft tissue fixation in the face
with the use of a micro mitek anchor. J Craniofac Surg 2005;16:117119
3. Okazaki M, Haramoto U, Akizuki T, et al. Avoiding ectropion by using
the Mitek anchor system for flap fixation to the facial bones. Ann Plast
Surg 1998;40:169173
Primary Giant Echinococcosis

of the Neck
Ozan Kuduban, MD, Afak Durur Karakaya, MD,y
Harun Ucuncu, MD,z and Muhammed Sedat Sakat, MDz
Abstract: The authors report an isolated echinococcosis in a
17-year-old girl presented with swelling of right neck approximately
12 cm in diameters with no evidence of the disease elsewhere in the
body. A thorough search of the literature revealed only a few cases of
isolated cervical echinococcosis. Surgical procedure was planned for
our case with the guidance of the magnetic resonance imaging, which
showed right cystic mass and within a laminar membrane. Histopathologic report confirmed echinococcosis. In summary, this current
study shows that in the differential diagnosis of the cystic masses
localized in the neck in the endemic regions, echinococcosis should
be suggested. In these patients, the careful assessment of the magnetic
resonance imaging evaluation before the surgery would extremely
facilitate either the diagnosis or the surgery planning.
Key Words: Echinococcosis, hydatid cyst, neck, parasitic
infestation, primary
chinococcosis is one of the zoonotic disease, which has large

geographical spread in the world.1 A thorough search of the
From the Department of Otolaryngology Head and Neck Surgery;

yDepartment of Radiology, Erzurum Regional Education and Research
Hospital; and zDepartment of Otolaryngology Head and Neck Surgery,
Ataturk University Medical Faculty, Erzurum, Turkey.
Address correspondence and reprint requests to Ozan Kuduban, MD,
Department of Otolaryngology Head and Neck Surgery, Erzurum
Regional Education and Research Hospital, 25341 Erzurum, Turkey;
E-mail: ozankuduban@gmail.com
The authors have no conflicts of interest.
ISSN: 1049-2275
DOI: 10.1097/SCS.0000000000002316
#
FIGURE 1. A, Preoperative T2-weighted coronal and sagittal neck magnetic

resonance imaging. B, Perioperative view of excised echinococcosis and its
laminar membrane.
literature revealed only a few cases of isolated cervical echinococcosis.2 We report an echinococcosis in a patient presented at right
neck with no evidence of the disease elsewhere in the body.
A 17-year-old girl was consulted to our outpatient clinic because
of the swelling growing at the right side of the neck that she had
noted for 2 months. There was a painless mass with soft stiffness in
12 10 in diameters at the right supraclavicular region. She had no
other systemic disease in her medical history. T2-weighted coronal
image of magnetic resonance imaging of neck showed a cystic
view containing linear appearances without signal belonging to
laminar membrane (Fig. 1A). The mass was excised under general
anesthesia. During the surgery, the laminar membrane emerged after
the capsule of cystic mass extending toward the bottom of the clavicula
was ruptured and this was excised separately from the specimen
(Fig. 1B). Albendazol 800 mg/d was begun on the postoperative first
day in 2 equal doses after consulting the infectious disease clinic.
No recurrence was seen in the follow-up of the patient.
The agent of echinococcosis is the larva in the metacestode
stage of Echinococcus granulosus, which is contaminated through
oralfecal route.3 Clinical signs appear with compression effect
depending on the involvement of organ placed in the tissue. In our
case, giant cystic mass, which reached 12 cm being localized in the
neck with fast-growing history suggested first to be a congenital
neck mass. Radiology helped us to establish the diagnosis and it
contributed much us to plan the treatment of echinococcosis.4
Although ultrasound examination determines only the nature of
the cervical mass as solid or cystic, computed tomography demonstrates cystic structure with multivesicular lesions. By this sign of
the computed tomography, the patient could be misdiagnosed as
pyogenic abscess or tuberculosis and this might lead draining the
lesion externally or administrating antituberculosis treatment. After
all, the assessment of magnetic resonance imaging for echinococcosis can allow physician to reach absolute conclusion about the
disease by determining laminar membrane.
There is no serologic test with sufficient specificity for the
diagnosis of echinococcosis. Surgical excision is the main treatment, which can be decided with clinical evidence and radiologic
examinations.5 Although unblock removal of the cyst is valuable,
cysts with deeper localizations are more prone to be ruptured. It
should be washed with protoscolicidal solutions. Because it will be
useful in preventing postoperative recurrence besides helping to
surgical treatment, anthelmintic medication is important.6
In conclusion, echinococcosis should be considered in the
differential diagnosis of the cystic masses localized in the neck
in the endemic regions by otolaryngologists.
REFERENCES
1. Sultana N, Hashim TK, Jan SY, et al. Primary cervical hydatid cyst: a rare
occurrence. Diagn Pathol 2012;7:157
2. Pratima K, Pooja K, Renu G, et al. Isolated echinococcosis of cervical
region. J Cytol 2014;31:102104
3. Macchiaroli N, Cucher M, Zarowiecki M, et al. microRNA profiling in
the zoonotic parasite Echinococcus canadensis using a high-through put
approach. Parasit Vectors 2015;8:83
4. La Plante JK, Pierson NS, Hedlund GL. Common pediatric head and neck
congenital/development abnormalities. Radiol Clin North Am
2015;53:181196
e101
5. Djuricic SM, Grebeldinger S, Kafka DI, et al. Cystic echinococcosis in

children: the seventeen-year experience of two large medical centers
in Serbia. Parasitol Int 2010;59:257261
6. Hemphill A, Stadelmann B, Rufener R, et al. Treatment of echinococcosis:
albendazole and mebendazole: what else? Parasite 2014;21:70
Components of Patient
Satisfaction After
Orthognathic Surgery
Kenneth Kufta, BS, Zachary S. Peacock, DMD, MD,y
Sung-Kiang Chuang, DMD, MD,y Gino Inverso, DMD,z
and Lawrence M. Levin, DMD, MD
Abstract: The purpose of this study was to compare overall patient
satisfaction after orthognathic surgery with the following specific
categories: appearance, functional ability, general health, sociability, and patientclinician communication. A 16-question survey
was developed and administered to include patients at either 6 or
12 months after orthognathic surgery between June 2013 and June
2014 at the University of Pennsylvania and Massachusetts General
Hospital. The predictor variables included age, sex, type of procedure, medical comorbidities, intra- or postoperative complications, and presence of paresthesia. The outcome variable was
patient satisfaction overall and in each category based on a Likert
scale (0: not satisfied at all to 5: very satisfied).
A total of 37 patients completed the survey and had a high
overall rate of satisfaction (100% of responses were 4 or 5 on Likert
scale). Overall satisfaction had the highest correlation with appearance (r 0.52, P 0.0009) followed by sociability (r 0.47,
P 0.004), patientclinician communication (r 0.38, P 0.02)
functionality (r 0.19, P 0.26), and general health (r 0.11,
P 0.51). Patients had high satisfaction scores for orthognathic
surgery. Satisfaction with postoperative appearance had the strongest correlation with overall satisfaction.
Key Words: Bilaterally sagittal split osteotomy, Le Fort I
osteotomy, orthognathic surgery, patient satisfaction
entofacial deformities can significantly impact quality of life.

Fortunately, orthognathic correction results in patient satisfaction rates of greater than 80%.1,2 Successful surgical outcomes
From the Harvard School of Dental Medicine; yOral and Maxillofacial

Surgery Service, Harvard Dental Center, Massachusetts General Hospital, Boston, MA; zDepartment of Oral and Maxillofacial Surgery,
Hospital of the University of Pennsylvania; and Division of Oral
and Maxillofacial Surgery, Childrens Hospital of Philadelphia, University of Pennsylvania; Division of Dentistry, Philadelphia, PA.
Address correspondence and reprint requests to Zachary S. Peacock, DMD,
MD, Oral and Maxillofacial Surgery, Mass General Hospital and Harvard School of Dental Medicine, 55 Fruit Street, Warren Building, Suite
1201, Boston, MA 02114; E-mail: zpeacock@partners.org
ISSN: 1049-2275
DOI: 10.1097/SCS.0000000000002318
e102
depend not only upon restoring a functional occlusion and aesthetic

facial harmony, but also patient satisfaction.3,4 It has been well
documented that there are various factors that determine overall
patient satisfaction.2,3,5
The patients assessment of facial appearance, occlusion, speech
articulation, relief of jaw pain, and self-confidence are known
variables that affect the patient-perceived treatment outcome.2,3,6
Appearance is most often cited as the strongest motive to undergo
surgery, and favorable aesthetic outcomes lead to more highly
satisfied patients.4,7 The amount of pre- and postoperative information provided as well as the effective surgeonpatient communication regarding the operation and recovery have been linked
to patient satisfaction.6,8 Despite overall success, inherent risks and
complications of orthognathic surgery can decrease patient satisfaction.1,2,9
Phillips et al10 have reported that patients tend to exhibit
dissatisfaction when they experience decreased bite strength,
difficulty chewing, numbness, and more postoperative facial
swelling than expected. Patients were more likely to exhibit
dissatisfaction pertaining to these elements of recovery than facial
appearance or social interaction.10 Ineffective communication as
well as inadequate information regarding postsurgical risks can also
result in patient dissatisfaction.1,8
Patient dissatisfaction can have significant psychosocial
effects.11,12 Schmidt et al11 have shown that patients who have
undergone orthognathic surgery exhibit a lower oral health related
quality of life based on functional, aesthetic, psychologic, and
social outcomes. Thus, patient satisfaction studies are important
as they can identify areas of improvement and allow tailoring of
communication and treatment to match expectations.
Patients are nearly 8 times more likely to be satisfied when
others have noticed a change in appearance.7 Few other data
exist on what particular changes are related to overall satisfaction. The purpose of this study was to compare overall patient
satisfaction with specific categories of satisfaction: appearance,
functionality, general health, sociability, and patient-clinician
communication after orthognathic surgery. We hypothesized that
appearance would be the strongest determinant of overall patient
satisfaction.

Study Design
Potential patients were determined from the department surgical
logs and billing data at The Hospital of the University of Pennsylvania and Massachusetts General Hospital. Patients that had orthognathic surgery [Le Fort I osteotomy, bilateral sagittal split
mandibular osteotomy (BSSO), or both] that presented for 6- or
12-month postoperative follow-up appointments were included.
Patients with craniofacial syndromes and those undergoing distraction osteogenesis or condylar reconstruction were excluded.
Included patients were provided with detailed information about
the study and informed consent was obtained before participation.
The study was approved by the Institutional Review Board at the
Hospital of the University of Pennsylvania (protocol number:
817652) and subsequent review was ceded by the Massachusetts
General Hospital Institutional Review Board.
Satisfaction Survey
A 16 question survey was designed to comprehensively
assess patient satisfaction. After assessing overall satisfaction, each
question was placed into 1 of 5 categories: appearance, functional
abilities, general health, sociability, and patientclinician communication (Table 1).
#
TABLE 1. Sixteen Questions Administered to Patients at 6-Month or 1-Year

Follow-Up Visit
TABLE 2. Summary of Study Variables

Study Variable
1. In general, how satisfied are you with the overall results of your surgery?
2. How satisfied are you with your current speech articulation?
3. How satisfied are you with your current lip closure?
4. How satisfied are you with your ability to bite/chew?
5. How satisfied are you with your current facial/jaw pain?
6. How satisfied are you with your current bite/occlusion?
7. How satisfied are you with the overall appearance of your teeth?
8. How satisfied are you with your ability to swallow?
9. How satisfied are you with your ability to breath?
10. How satisfied are you with your ability to sleep?
11. How satisfied are you with your general health?
12. How satisfied are you with your general appearance?
13. How satisfied are you with your ability to socialize in public?
14. How satisfied are you with your interpersonal relationships?
15. How satisfied are you with your preoperative counseling?
16. How satisfied are you with your postoperative counseling?
Sample size (N)

Mean age (years)
Sex: women
Le Fort I Genioplasty
BSSO Genioplasty
Le Fort I BSSO
Le Fort I BSSO Genioplasty
Subject survey data were imported into an Excel (Microsoft,
Redmond, WA) spreadsheet and analyzed using Statistical Analysis
System and Statistical Analysis System Version 9.3 (Cary, NC).
Descriptive statistics were computed for all of the study variables and the Spearman correlation coefficients (r) were investigated to compare the relationship between study variables. A
correlational analysis was developed to identify study variables
that were associated with changes in overall satisfaction outcome.
37
23.5
57%
40.5%
32.4%
16.2%
10.8%
BSSO, bilateral sagittal split mandibular osteotomy.
TABLE 3. Number of Patients Reporting Postoperative Complications After

Orthognathic Surgery at 6-Month or 1-Year Follow-up Visits
Paresthesia
Infection
Sequestrum from proximal segment
Additional data collected in the survey included: age, sex, type

of procedure, current or former use of tobacco, medical comorbidities (ie, breathing disorder or obstructive sleep apnea, and any
psychiatric disorder). Follow-up data were analyzed to evaluate
evidence of postoperative paresthesia, infection, or necrosis. The
primary outcome variable was patient satisfaction in each category
based on a Likert scale (0: not satisfied at all to 5: very satisfied).
The mean score was calculated for each category and assessed based
on a semiquantitative scale as follows: 1 and 2 unsatisfactory,
3 neutral, and 4 and 5 satisfactory (Fig. 1).
Descriptive Statistics
6-Month (%)
1-Year (%)
10 (28)
0 (0)
1 (3)
5 (17)
0 (0)
0 (0)
TABLE 4. Mean Values and Standard Deviations for Survey Responses by

Category
Category
Overall
Functionality
Appearance
Sociability
General health
Patientclinician communication
Mean
4.92 0.28
4.84 0.09
4.84 0.16
4.86 0.04
4.79 0.06
4.85 0.02
A total of 37 patients (21 women) completed the survey. The mean

age of the sample was 23.5 years 10.9. Of the patients who
completed the survey, 40.5% (15 patients) underwent a Le Fort I
osteotomy with or without genioplasty, 32.4% (12 patients) underwent a BSSO with or without genioplasty, 16.2% (6 patients)
underwent Le Fort I and BSSO, and 10.8% (4 patients) underwent
Le Fort I, BSSO, and genioplasty (Table 2). Of the study sample, 4
patients reported tobacco use, 9 had a history of TMJ dysfunction, 7
had obstructive sleep apnea, and 5 had asthma. Postoperative
complications were also retrospectively evaluated at 6-month

and 1-year follow-up visits. Of the 37 patients, 36 had complete
records of 6-month postoperative data, while 30 had complete
records of 1-year follow-up data. At the 6-month follow-up, 10
patients reported paresthesia, 1 patient had undergone hardware
removal because of proximal segment necrosis post-BSSO, while
no patients showed signs of infection. At the 1-year follow-up, 5
patients reported paresthesia, while no patients showed signs of
bone necrosis or infection (Table 3).
All patients had high overall rate of satisfaction with all responses
4 (mean 4.92. 0.28) (Fig. 1, Table 4). Overall satisfaction had the
highest correlation with appearance (r 0.52, P 0.0009). Other
categories were correlated as follows: functionality (r 0.19,
P 0.26), general health (r 0.11, P 0.51), sociability
(r 0.47, P 0.004), and patient-clinician communication
(r 0.38, P 0.02) (Fig. 2). Spearman correlation coefficients were
also calculated to investigate the relationship between each of the
FIGURE 1. Mean values for answers to each survey question (37 responses total
for each question).
FIGURE 2. Spearman correlation describing relationship between overall

satisfaction with procedure and each of the study variables.
RESULTS
e103
TABLE 5. Summary of Spearman Correlation Coefficients Between Individual

Study Variables
Function Appearance General Sociability PatientDoctor
Health
Communication
Overall (Q1)
Function
Appearance
General health
Sociability
Patientdoctor
communication
0.19
1
0.52
0.19
1
0.11
0.35
0.31
1
0.47
0.16
0.52
0.24
1
0.38
0.05
0.51
0.28
0.69
1
5 categories (Table 5). There was no significant correlation between

demographical data or type of procedure and satisfaction outcomes.
DISCUSSION
Patient satisfaction with orthognathic surgery remains high. The
purpose of this study was to offer a more comprehensive and
categorical assessment of patient satisfaction. The study allowed
us to determine which aspects of satisfaction most strongly correlated with overall satisfaction and other study variables. We
hypothesized that appearance would most highly correlate with
overall satisfaction.7
Patients included in this study were highly satisfied with their
surgical experiences. All 37 patients reported a 4 or 5 on the Likert
scale when asked about overall satisfaction. These results are in
agreement with previous studies that show high (>80%) overall
rates of satisfaction with orthognathic procedures.1,2 As demonstrated in previous studies, our study showed that patients were
satisfied with improvements made in jaw function, facial appearance, and comfort level in social environments.1,2,13 Posnick et al2
in a similar study reported that 88% of patients reported satisfaction with outcome and 93% would recommend the surgery
to others.
Satisfaction with functionality (r 0.19) was less important to
overall satisfaction than appearance (r 0.52). Functional aspects
may have been minimized because of the indications for surgery being
strongly for aesthetic reasons or general health (eg, obstructive sleep
apnea). If patients did not have preoperative maxillofacial functional
concerns, it is not likely that any changes in postoperative functional
aspects would contribute greatly to their overall satisfaction.
The relationship of appearance to overall satisfaction was highly
significant (P 0.0009). The strong relationship between overall
satisfaction and that with appearance may be affected by the high
number of women in this study. Studies have shown that women are
more likely to seek orthognathic surgical correction while placing a
greater emphasis on aesthetic improvement.6,14,15
Sociability was also significantly correlated with overall satisfaction (r 0.47, P 0.004). Personality and self-esteem can be
significantly affected by the results of orthognathic surgery.16
Kiyak et al17 also showed that a decline in satisfaction with facial
body image 9 months after orthognathic correction results in a
similar decrease in both personal and social self-esteems. In our
study, it is likely that patients were more comfortable in a social
setting when satisfied with their postoperative appearance; the
relationship was highly correlated (r 0.52, P 0.001). Interestingly, satisfaction with general health negatively correlated with
overall satisfaction ratings. Perhaps patients with poor general
health or extraoral, systemic complications are more likely to report
overall satisfaction with their orthognathic procedure. An accurate
explanation would require more detailed investigation of the effect
of various medical comorbidities on the categories of satisfaction.
e104
Satisfaction with patientdoctor communication before and

after the operation was highly associated with overall satisfaction
(r 0.38, P 0.02), with appearance (r 0.51, P 0.001), and
sociability (r 0.69, P < 0.0001). This is in agreement with
previous studies on doctorpatient communication.6,8,18,19 Turker
et al18 showed that a thorough preoperative explanation of the
surgical procedure had a significant positive correlation with trust in
the surgical team and postoperative satisfaction. In addition, Flanary et al6 found that surgical complications and outcomes that were
not adequately discussed with patients had a major influence on
patient satisfaction. Thus, thorough and appropriate explanation of
the procedure, potential side effects, and complications may
increase overall satisfaction with the surgical result and recovery.12
There are several limitations of this study. Patients may be
hesitant to express dissatisfaction for fear of offending the surgeon,
which could have falsely elevated satisfaction scores. In addition,
many included patients were still wearing orthodontic appliances at
the time of survey completion, which could have affected the results
regarding social comfort. The limited sample size limits conclusions. A modified version of this survey will be used to study
a larger sample size as well as evaluate any association between
satisfaction and postoperative complications.
Overall satisfaction with orthognathic surgery is high. Overall
satisfaction has the highest correlation with that of appearance
followed by sociability and patientclinician communication.
REFERENCES
1. Al Kharafi L, Al Hajery D, Andersson L. Orthognathic surgery:
pretreatment information and patient satisfaction. Med Princ Pract
2014;23:218224
2. Posnick JC, Wallace J. Complex orthognathic surgery: assessment of
patient satisfaction. J Oral Maxillofac Surg 2008;66:934942
3. Modig M, Andersson L, Wardh I. Patients perception of improvement
after orthognathic surgery: pilot study. Br J Oral Maxillofac Surg
2006;44:2427
4. Ostler S, Kiyak HA. Treatment expectations versus outcomes among
orthognathic surgery patients. Int J Adult Orthodont Orthognath Surg
1991;6:247255
5. Travess HC, Newton JT, Sandy JR, et al. The development of a patientcentered measure of the process and outcome of combined orthodontic
and orthognathic treatment. J Orthod 2004;31:220234
6. Flanary CM, Barnwell GM, Alexander JM. Patient perceptions
of orthognathic surgery. Am J Orthod 1985;88:137145
7. Trovik TA, Wisth PJ, Tornes K, et al. Patients perceptions of
improvements after bilateral sagittal split osteotomy advancement
surgery: 10 to 14 years of follow-up. Am J Orthod Dentofacial Orthop
2012;141:204212
8. Levinson W, Hudak P, Tricco AC. A systematic review of surgeon
patient communication: strengths and opportunities for improvement.
Patient Educ Couns 2013;93:317
9. Kim SG, Park SS. Incidence of complications and problems related to
orthognathic surgery. J Oral Maxillofac Surg 2007;65:24382444
10. Phillips C, Kiyak HA, Bloomquist D, et al. Perceptions of recovery and
satisfaction in the short term after orthognathic surgery. J Oral
11. Schmidt A, Ciesielski R, Orthuber W, et al. Survey of oral health-related
quality of life among skeletal malocclusion patients following
orthodontic treatment and orthognathic surgery. J Orofac Orthop
2013;74:287294
12. Hunt OT, Johnston CD, Hepper PG, et al. The psychosocial impact of
orthognathic surgery: a systematic review. Am J Orthod Dentofacial
Orthop 2001;120:490497
13. Dantas JF, Neto JN, de Carvalho SH, et al. Satisfaction of skeletal class
III patients treated with different types of orthognathic surgery. Int J
Oral Maxillofac Surg 2015;44:195202
14. Kiyak HA, McNeill RW, West RA. The emotional impact of
orthognathic surgery and conventional orthodontics. Am J Orthod
1985;88:224234
#
15. Peacock ZS, Lee CY, Klein KP, et al. Orthoghnathic surgery in oatients
over 40 years of age: indications and special considerations. J Oral
16. Kiyak HA, West RA, Hohl T, et al. The psychological impact of
orthognathic surgery: a 9-month follow-up. Am J Orthod 1982;81:
404412
17. Kiyak HA, Hohl T, West RA, et al. Psychologic changes in orthognathic
surgery patients: a 24-month follow up. J Oral Maxillofac Surg
1984;42:506512
gel K, et al. Perceptions of preoperative
18. Turker N, Varol A, O
expectations and postoperative outcomes from orthognathic surgery:
part I: Turkish female patients. Int J Orthognath Surg 2008;37:710715
19. Peterson LJ, Topazian RG. The preoperative interview and
psychological evaluation of the orthognathic surgery patient. J Oral
Surg 1974;32:583588
Incomplete Reossification After

Craniosynostosis Surgery
Niels Noordzij, MD, Roma Brouwer, MD,y and
Chantal van der Horst, PhD
Abstract: A patient with unicoronal craniosynostosis was treated
by an open cranial vault remodeling procedure at 11 months of age.
A calvarial defect persists at the site of the sagittal suture at 7 years
follow-up. This unexpected outcome led us to evaluate current
literature on incidence and possible causes of incomplete reossification after craniosynostosis surgery.
English literature was searched from 1982 to 2013. Variables of
interest were incidence, diagnose, type of surgery, age at operation,
possible causes for incomplete reossification, and duration and type
of follow-up.
Incidence in unselected cohorts ranged from 0.5% to 18.2%.
Incomplete reossification has been reported in syndromical and
nonsyndromical cases, after multiple types of surgery for any type
of suture. Follow-up was done by palpation, radiology or both, for a
period of 6 to 264 months.
Higher age at operation and a dura tear are associated with an
increased risk of incomplete reossification.
Type and duration of follow-up is inhomogeneous and there is a
wide variety in the terminology used for incomplete reossification.
To improve international communication on this topic, more
long-term studies, using a consistent type and length of follow-up
and uniform terminology are needed.
urgical correction of isolated craniosynostosis aims at prevention of functional impairment because of raised intracranial
pressure, but mainly at achieving an aesthetic satisfactory shape.1 3
Multiple techniques have been described and practiced for the
correction of craniosynostosis, depending on the suture(s) affected
and age of presentation.312 In many techniques, the skull is left not
completely covered with bone, for the patient will grow new bone in
these gaps. Studies have shown the dura to be the main source of
new bone in this reossification process.13 16 We report a case where
this process of reossification did not take place.
PATIENT
A girl with unilateral coronal craniosynostosis was operated on at
the age of 10 months using combined open strip craniectomy and
cranial vault remodeling.
Follow-up was done using plain radiographs and by palpation.
She showed a lack of reossification of the osseous gap left at surgery
(Fig. 1), still present at 7 years follow-up (Fig. 2). No literature was
found on how to treat such a patient. Her parents often worried
about the (unknown) consequences, but she showed no physical
complaints of the lack of reossification. Therefore, management
stayed conservative.
This case stimulated us to perform an evaluation of the literature
on incomplete reossification (or residual calvarial defects) after
craniosynostosis surgery and look into its incidence and possible
causes.
METHODS
PubMed and Cochrane databases were searched using (combinations of) the following search terms: (isolated/nonsyndromic)
craniosynostosis, surgery, (long term) results, age at surgery, bony
defect, calvarial defect, cranial defect, bony gap, osseous defect,
growing skull fracture (GSF), complications, dura tear, (incomplete) reossification, follow-up.
English literature from 1982 to 2013 was searched. We included
any type of article that reported cases of incomplete reossification.
Variables of interest were incidence of incomplete reossification/residual calvarial defects, diagnosis, surgical technique, age at
operation, duration and way of follow-up and suggested causes
described.
Key Words: Calvarial defect, complications, craniosynostosis,

dura, follow-up, reossification, surgery
FIGURE 1. Radiograph 6 months after surgery.
From the Academic Medical Center, Amsterdam; and yMedical Center

Alkmaar, the Netherlands.
Address correspondence and reprint requests to Niels Noordzij, MD,
Academic Medical Center, Oudezijds Achterburgwal 82, 1012 DR
Amsterdam, the Netherlands; E-mail: nnoordzij@gmail.com
The authors report no conflict of interest.
ISSN: 1049-2275
DOI: 10.1097/SCS.0000000000002319
FIGURE 2. Radiograph 7 years after surgery.
e105
RESULTS
16 studies were found reporting incomplete reossification in their
patients. (Table 1) Reported incidence in unselected cohorts ranged
between 0.5%17 and 18.2%18 The occurrence of incomplete reossification after craniosynostosis surgery is reinforced by several
case reports.19
Incomplete reossification is reported in syndromic20 and nonsyndromic cases,21 after all types of surgery,21,22 and after treatment of
any suture.21 Mean age at surgery varied from 3 to 47 months.23,24
Follow-up varied from 6 to 264 months after surgery and was done
either by palpation,20,25 using radiology16,26 or both.21,27 (Table 1) In
several studies, the type of follow-up was unclear.5,17,24,28 Higher age
of the patient at surgery is associated with incomplete reossification,7,16,20,24,2934 but only a few studies have systematically
examined and suggested its relation.16,35,36 All 3 show significantly
more incomplete reossification in patient groups operated on at
12 months or older. Also, the presence of a defect of the dura is
described as a cause for incomplete reossification.3,19,29,30,3740
DISCUSSION
Incidence and Follow-Up
It is hard to draw conclusions from the results of incidence rates
as presented in Table 1, because this list comprises retrospective
studies that differ in diagnosis, age at surgery, operation types and
way and duration of follow-up, even within a single article.5,24,28
Several reasons might explain why this list is short in number and
why the variation in incidence rates is high:
1. The terminology used to describe incomplete reossification is
highly variable. Bony defect,41 cranial lacuna,5 residual cranial
defect,8 persistent calvarial defect,12 persistent osseous defect,20
and residual osseous defects42 all indicate the same situation.
Also, GSF after surgery,3,38,43,19 encephalocele,28 pseudomeningocele,22,44 and leptomeningeal cyst,40 atrogenic intradiploic meningoencephalocele after craniosynostosis surgery45
are different names to describe the closely resembling situation of
incomplete reossification resulting from a dura defect. Variable
terminology makes it difficult to easily acquire and compare
literature. Hence, we insist on a more consistent terminology.
2. Follow-up varies greatly between centers. There is no uniform
protocol, which make outcomes hard to compare. Plain
radiographs are used to evaluate suture morphology and
patency,26 but reossification is not evaluated well on static twodimensional imaging. Computed tomography scans provide
better images,16 but cause extra radiation and often require a
form of anaesthesia. Others apply only palpation to evaluate
reossification,20,36 arguing that defects not detectable by
palpation are clinically irrelevant. What defect size might still
be detected by palpation is not mentioned. Also, what factors
influence the decision to repair a calvarial defect are not
addressed. For incidence rates, it is essential to report to actively
have searched for calvarial defects. Paige36 used the reported
presence or absence of a palpated bony defect and a follow-up
duration of at least 12 months as inclusion criteria, leading to
inclusion of only 81 of 347 operated patients in their study.
(Table 1) Patients with bony defects are followed more closely,
leading to the report of that defect in their file. This might
explain their high incidence rate.36 A standardized way of
follow-up will greatly improve the possibilities to evaluate and
compare surgical therapy. Duration of follow-up varies as well.
Reossification time is dependent on age at surgery,16,29 where
older children take longer to reossify their defects than younger
children. Therefore, follow-up in older children should
emphasize longer on reossification than in younger children.
Paige36 mentioned that no calvarial gaps that were present after
TABLE 1. Incidence and Follow-Up of Incomplete Reossification

Pts With
Bony
Defect(s)
Publication
Pts in Study
32
Whitaker 1987
Hassler 199020 [20]
Hudgins 199330
Prevot 199323
Burstein 199448
Moss 199533
Wagner 199526
Paige 200629
Jimenez 200724
Greene 200722
Selber 200839
Esparza 200810
Seruya 201134
Seruya 201145
Rottgers 201131
Jimenez 201359
164
60
9
592
18
42(a)
22
81
100
213
81
283
212
17
10
115
Mean Age
at Surgery
Type of Synostosis Includes Syndromic
2
1.2 Various
4
6.7 Sagittal
1 11.1 Sagittal
30 5.1 Various
1
5.6 Sagittal
42 100.0 Various
4 18.2 Bicoronal
58 71.6 Various
2
2.0 Metopic or coronal
21 9.9 Various
5
6.2 Unilateral coronal
7
2.5 Various
1
0.5 All single sutures
2 11.8 Sagittal
6 60.0 Sagittal
3
2.6 (Bi-)coronal
Yes
No
No
Yes
No
Yes
No
Yes
No
Yes
No
Yes
Yes
No
No
No
Type of Surgery
(Months)
Multiple
Open, vault remodeling
Multiple
Multiple
Endoscopic, strip craniectomy
Multiple
Multiple
Endoscopic, strip craniectomy
Follow-Up
12119
1260
9
12228
36
Max 36
6168
12
60
36264
69
1560d
19
12
47
6
24
4
272d
11
7
11
41
32
3
Follow-Up Type
f
Radiology
Palpation
Palpation radiology
f
f
Palpation radiology
Palpation
Palpation
Palpation
Palpation
0.5138
0.692
3112
6144
f
f
Palpation radiology
f
Thirty three of 42 where craniosynostosis patients. A bony defect was an inclusion criterion for this study.
Distribution of patients was: <1.5 years (N 92), >1.5 years (N 72). Mean age was not mentioned and could not be calculated.
Distribution of patients was: <6 (N 42), 7 to 12 (N 14), >12 (N 4). Mean agewas not mentioned and could not be calculated.
d
Mean age could not be calculated.
e
Duration of follow-up not mentioned in article.
f
Type of follow-up unknown (not mentioned in article).
b
c
e106
1 year subsequently closed completely, but their patient group

consisted mainly of syndromic synostosis patients, with age at
operation of 23.9 28.6 months (median age 12 months).
Hassler16 showed reossification times up to 18 months in
patients operated on between 7 and 12 months of age.
5
3. Topic not of interest: Esparza reports 7 patients of 283
craniosynostosis patients with persistent cranial defects. These
were named irrelevant, although the consequence was
another surgical procedure. No further details are mentioned.
Ferreira4 excluded 6 patients for their atypical timing of
treatment (average age 72 months) in their study reviewing
the surgical treatment of nonsyndromic craniosynostosis.
Jimenez22 reports 2 dura tears and 4 pseudomeningoceles in
an analysis of 100 minimally invasive endoscopic-assisted
suturectomies. There were 2 patients of incomplete reossification, but the article focuses on morphologic outcomes and
perioperative complications. The reason for incomplete reossification or its consequence is not addressed. More authors sharing
this view will lead to under documentation of the subject.
Increasingly, the result of therapy is evaluated by Patient Reported
Outcome Measures.46 In our list of studies, no mention is made about
patient (parents) opinions in case of incomplete reossification. This
complication caused great concern in the parents of our patient and
this should be included and reported in follow-up.
Reasons for Incomplete Reossification

Age at Surgery
Animal studies reinforce the relationship between age at
operation and closure of calvarial defects.13,15,47,48
There, however, is no consensus at what age this reossification
potential becomes critical. Authors state critical ages of 9,42 10,32
12,7,31,3518,24,34 and even 24 months.25,29,33
Interestingly, Prevot et al21 could not link age at initial surgery
as cause for persistent calvarial defects in their extensive evaluation
on this subject. Two authors who studied reossification as a function
of age at surgery concluded reossification was incomplete at final
follow-up in patients operated on at ages older than 12 months.16,36
All conclude to perform elective surgery for single suture synostosis
at the latest before 12 months of age. This is in accordance with
literature on preferred timing of elective craniosynostosis surgery of
many craniofacial centers, where patient age is between 3 and
12 months depending on the type of suture affected and type of
surgery used.7 9,29,32,49,50 Wagner18 and Selber35 also suggest
patient age as the cause for their patients requiring reoperation
for persistent calvarial defects (Table 1).
Unfortunately, patient referral may still happen at ages beyond
preferred surgical correction.7,8,25,27,41,51,17 In the case of late
referral, it is essential for the surgical team to know whether it
can rely on spontaneous reossification. In fact, many centers do not
leave reossification to chance and fill the bony gaps with some sort
of material.20,29,31,32,52 Rottgers et al27 reviewed the outcomes of 10
patients operated on at a mean age of 1.75 years and reported a 60 %
incidence of cranial defects (N 6) at final follow-up even with
filling of the bony gaps, (Table 1) although they judged the defects
only visible on computed tomography not significant. Hudgins25
and Seruya also filled the defects with graft material and report
incidence rates of 11.11 and 11.76 %, respectively, but their patient
groups were also small (Table 1).
Dura Tear
The bone regenerating capacities of the dura have been
established in a series of animal studies.13 16
Integrity of the dura is obligatory: the situation of a dura
defect after craniosynostosis surgery may cause a GSF as after
#
trauma.3,19,30,3740 Fourteen of these patients were presented by

Zemann.19
Risk factors for a dura tear include: the dura in infants is tightly
adherent to the under surface of bone at the suture lines,37,39
reoperation, because of adhesions formed after initial surgery,5,19
and raised intracranial pressure.19 Some authors warn for the
potential of missing a dura defect because of its limited exposure,
for the most probable site of injury of the dura exactly underlies the
bone edge.37 Given the potential of a bony defect to occur after
primary closure of a dura defect even with well-placed sutures,3 we
recommend proper documentation of every dura laceration during
surgery and specific attention during follow-up.
Whether endoscopically assisted surgery is a risk factor for
missing a dura tear is still under debate.3,10,19,40,53
Defect Size
Whether the size of the calvarial defect left at surgery has
consequences for its closure, remains a topic of discussion. There
are authors who say size does24 and who say size does not
matter.13,16,36 Regarding an intact dura to be the source of central
new calvarial bone16,48 this ought to be a manageable obstacle in
patients with a potent dura, even in big calvarial defects.16,54
Several authors state that the critical size of a defect is negatively
correlated with patient age.31,42 This might explain why our patient
failed to completely reossify her defect.
Infection
The negative effect of wound infection on closure of the
calvarial defect has been addressed, but its relation proves hard
to be made significant, probably because its incidence after craniosynostosis surgery is low.9,16,17,22,23,31,32,35,36
Looking at our case, no tear of the dura was noted and there was
no infection. Age at surgery was within the preferred age at surgery
range. We hypothesize that the size of the gap left open at surgery
was too big for her age to completely reossify, but we lack an
evidence based explanation.
CONCLUSIONS
Literature regarding incomplete reossification after craniosynostosis surgery is scarce and heterogeneous regarding incidence, terminology and follow-up. Reported incidence in unselected cohorts
ranged between 0.5 and 18.2 %. Adequate calvarial reossification
seems to depend heavily on the bone regenerating capacity of the
dura. This requires the dura to be intact and this capacity decreases
with age. The discrepancy between authors at what age this dura
function begins to cease is striking. Best available evidence
suggests increased risk of incomplete reossification in patients
operated on at 12 months of age or older.
To improve international communication regarding diagnosis,
incidence, and management of incomplete reossification more
reports on long-term outcome of craniosynostosis surgery are
needed, using a more consistent terminology, type, and duration
of follow-up. We therefore urge more authors to report their longterm surgery outcomes, including the presence or absence of
residual calvarial defects.
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12. David LR, Plikaitis CM, Couture D, et al. Outcome analysis of our first
75 spring-assisted surgeries for scaphocephaly. J Craniofac Surg
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13. Reid CA, McCarthy JG, Kolber AB. A study of regeneration in parietal
bone defects in rabbits. Plast Reconstr Surg 1981;67:591596
14. Mossaz CF, Kokich VG. Redevelopment of the calvaria after partial
craniectomy in growing rabbits: the effect of altering dural continuity.
Acta Anat (Basel) 1981;109:321331
15. Hobar PC, Schreiber JS, McCarthy JG, et al. The role of the dura in
cranial bone regeneration in the immature animal. Plast Reconstr Surg
1993;92:405410
16. Hassler W, Zentner J. Radical osteoclastic craniectomy in sagittal
synostosis. Neurosurgery 1990;27:539543
17. Seruya M, Oh AK, Boyajian MJ, et al. Long-term outcomes of primary
craniofacial reconstruction for craniosynostosis: a 12-year experience.
Plast Reconstr Surg 2011;127:23972406
18. Wagner JD, Cohen SR, Maher H, et al. Critical analysis of results of
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Surg 1995;6:3237; discussion 389
19. Zemann W, Metzler P, Jacobsen C, et al. Growing skull fractures after
craniosynostosis repair: risk factors and treatment algorithm.
J Craniofac Surg 2012;23:12921295
20. Greene AK, Mulliken JB, Proctor MR, et al. Primary grafting with
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21. Prevot M, Renier D, Marchac D. Lack of ossification after cranioplasty
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following surgery for isolated sagittal craniosynostosis. J Neurosurg
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of the timing, treatment, and complications in 164 consecutive patients.
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fronto-orbital reconstruction in nonsyndromic unicoronal synostosis.
Plast Reconstr Surg 2008;121:251e260e
36. Paige KT, Vega SJ, Kelly CP, et al. Age-dependent closure of bony
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vault reshaping in infancy. J Craniofac Surg 1998;9:73 75
40. Aryan HE, Meltzer HS, Gerras GG, et al. Leptomeningeal cyst
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43. Muhonen MG, Piper JG, Menezes AH. Pathogenesis and treatment of
growing skull fractures. Surgical neurology 1995;43:367373
44. Elias DL, Kawamoto HK. Pseudomeningocele: an unusual complication
of craniofacial surgery. Plast Reconstr Surg 1992;90:484486
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calvarium in young normal and growth hormone-treated
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48. Gosain AK, Santoro TD, Song LS, et al. Osteogenesis in calvarial defects:
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adult versus infant animals. Plast Reconstr Surg 2003;112:515527
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#
Correction of Unilateral
Congenital ZygomaticoMandibular Fusion
James Rough, MD, David I. Hindin, MD,y
Justine C. Lee, MD, PhD, and James P. Bradley, MDy
Abstract: Sygnathia, or fusion of the jaw, is a rare condition in
children, occurring either in isolation or as part of a larger overall
syndrome. Consequences of this bony fusion may range from
feeding difficulties to a complete inability to protect the airway.
Owing to the uncommon nature of this problem and the high
recurrence of bony fusion, standardized treatment protocols do
not yet exist, making individual reports particularly useful for
guiding the first-time management of such patients. In this report,
we describe the case of a male infant with complete bony fusion of
the right zygomatic maxillary complex to the mandible. Fusion was
separated by osteotomy, repair of soft tissue with acellular dermal
matrix/grafting, and plate separation. Serial jaw manipulation and
operative stretching was necessary to prevent refusion of syngnathia
even in the long term.
Key Words: Synechiae, syngnathia, zygomatico-mandibular
synostosis
ongenital fusion of the maxilla and the mandible is a rare

anomaly. Patients may vary in severity from fusion limited to
the soft tissues (synechiae) to a presentation as extensive as bony
fusion (syngnathia).1 Syngnathia may present as an isolated finding,
although it may also be found in syndromes such as Van der Woude
Syndrome.2 Syngnathia is associated with neonatal problems,
including difficulty with airway maintenance and protection, and
the inability to feed. Although syngnathia involving fusion of the
alveolar ridges of the maxilla and the mandible has been known to
occur, fusion of the zygomaticomaxillary-mandibular regions is
extremely rare (less than 10 patients reported in the literature).3
Presently, we document the treatment of a newborn with zygomatico-mandibular synostosis in conjunction with an omphalocele and
an atrial septal defect.
Clinical Report
A male infant, born via C-section to a 34 year old G3P2 mother
at 39 5/7 week gestation, was noted to have facial asymmetry and
inability to open his mouth at birth. Appearance, Pulse, Grimace,
Activity, Respiration scores were 8 and 9. Initially, the patient did
not require mechanical ventilatory support and was transferred from
From the Division of Plastic & Reconstructive Surgery, University of
California, Los Angeles (UCLA), David Geffen School of Medicine,
Los Angeles, CA; and yDivision of Plastic & Reconstructive Surgery,
Temple University Hospital, Philadelphia, PA.
Address correspondence and reprint requests to James P. Bradley, MD,
Temple University Hospital, 3401N. Broad St, 4th Floor Parkinson
Pavilion Bldg., Suite 450, Philadelphia, PA 19140;
E-mail: JPBradley4@mac.com
ISSN: 1049-2275
DOI: 10.1097/SCS.0000000000002339
#
FIGURE 1. Newborn patient showing right lower face hypoplasia and cant with
feeding tube.
an outside institution, unintubated. On physical examination, he

was found to have right lower face hypoplasia, a cant, and fusion of
the right alveolar ridge, beginning just right of the midline and
extending back toward the posterior aspect of the gingiva (Fig. 1). In
addition, he demonstrated a moderate size central abdominal
omphalocele and a grade III heart murmur. Routine laboratory
workup was normal. Two-dimensional echocardiography demonstrated an atrial septal defect. Additional workup demonstrated
immature retinal vascularization. No other abnormalities were
noted. A Three-dimensional computed tomography scan of the
face demonstrated bony fusion of the right zygomatic maxillary
complex to the mandible (Fig. 2). Fusion extended anteriorly from
the incisor maxillomandibular region along the alveolar ridge
posteriorly up to, but not including, the temporomandibular joint.
The temporomandibular joint had a narrow joint space but otherwise appeared normal in shape and position. The orbits were
asymmetric in size and shape. The nasal root and chin point were
deviated to the right.
On postnatal day 5, the patient was brought to the operating
room with the primary concerns of airway protection and the need
for nutritional support. In addition, care was directed toward
correction of the omphalocele and correction of maxillomandibular
fusion. Fiberoptic guidance was used for initial nasal intubation. A
tracheostomy was performed, followed by gastrostomy tube placement and omphalocele repair. For correction of right jaw fusion, we
began with a Risdon incision after local injection. Subperiosteal
dissection identified the region of abnormal bony fusion. The
condyle, angular notch, and coronoid process were present. The
coronoid process was high-riding, cephalad to the incomplete
zygomatic arch. Temporalis muscle had normal insertion to
the coronoid process. Intraorally, we found complete fusion of
the maxillomandibular gingiva and alveolus. We incised the buccal
gingiva and lingual gingiva with electrocautery, and did limited
subperiosteal undermining to expose the fused bones. Through the
FIGURE 2. Three-dimensional computed tomography images demonstrating

bony fusion of right zygomatic maxillary complex to the mandible, with
extension from right incisors up near the temporomandibular joint. A, Frontal
view. B, Right oblique view. C Right lateral view.
e109
for 1 month. He subsequently underwent 3 additional rounds of

jaw manipulation at 2, 6, and 9 months after his distractor
placement without any evidence of bony fusion. At 2.5 years
follow-up patient had maintained separation of the upper and
lower jaw. Evidence of growth was seen in the mandible with
partial correction of occlusal cant and chin point. Although
hypodontia existed in the involved alveolar regions, evidence
was seen of tooth bud development.
FIGURE 3. Intraoperative views. A, Frontal views shows right maxillomandibular
gingival and aveolar fusion. B, Right lateral view shows completion of osteotomy
through the Risdan incision.
Risdon incision, an osteotomy was performed with a reciprocating

saw whereas a malleable was used intraorally to protect the tongue
(Fig. 3). After complete separation, there was immediate movement
at the temporomandibular joint. Electrocautery was used to then
completely divide the remaining fused gingival mucosa. Thus, we
found good jaw mobility and did not find evidence of ankylosis after
separation. To prevent recurrence of bony fusion, we did a soft
tissue reconstruction over the osteotomy sites. We advanced local
soft tissue and placed 2 strips of acellular dermal matrix (approximately 2 cm 3.5 cm each), 1 over the maxilla alveolus and 1 over
the mandible alveolus. Grafts were inset using 40 chromic suture
in a simple interrupted fashion. We closed the external Risdon
incision in layers with interrupted 40 Monocryl and 50
Monocryl. The infant was transferred to the neonatal intensive
care unit in stable condition.
Postoperative management specific to our procedure included
jaw physiotherapy performed 10 times per hour while awake for
7 days. Subsequently, parents were instructed to perform daily
jaw exercises at least 5 times per day. Follow-up three-dimensional CT scan of the face confirmed separation and bony healing
(Fig. 4). Serial operative visits were used to stretch the soft tissues
every 6 weeks. At 4 months after the initial surgery, the jaws had
refused, potentially because of noncompliance with physical
therapy. He was then brought back to the operating room and
reosteotomized at the area of bony bridging. Alloderm was again
used to separate the maxilla from the mandible, and a KLS MicroZurich distractor was placed to hold the jaws open during healing
FIGURE 4. Three-dimensional computed tomography images demonstrating

separation of the maxilla and mandible. Bony fusion of right zygomatic
maxillary complex to the mandible. A, Frontal view. B, Right oblique view. C,
Right lateral view.
e110
DISCUSSION
Syngnathia is a rare condition with zygomaticomaxillary fusion
representing a small number of cases. A review of the literature
reveals less than 10 cases involving fusion of the zygomatic
complex.4 Early treatment is essential to allow for feeding, and
to protect the airway.5,6 Early treatment is also essential to facilitate
early jaw mobilization and prevent temporomandibular joint (TMJ)
ankylosis. Diagnosis of syngnathia is easily confirmed with CT scan
revealing the extent of bony fusion and joint involvement. This
information provides a roadmap for operative planning.
Congenital TMJ ankylosis can often lead to lifelong problems
for patients, often including severe difficulties with mouth opening,
inadequate nutritional intake, and even chronic hypoxia.7 When
bony ankylosis is noted at birth, the surrounding ligaments necessary for the translational component of TMJ function will remain
problematic through a patients life even with aggressive surgical
intervention and ongoing therapy. In the case described here,
correction of this patients zygomatic-maxillomandibular fusion
provided early use of the patients TMJ and full functional recovery.
With prolonged nonuse of the TMJ, full recovery would have been
less likely. It is also salient to note that the condyle remained
anatomically unaffected in this patients case. Previous studies have
shown that the condyle represents one of the most active bone
growth centers within the mandible.8 Growth for this patient has
been able to proceed in a normal fashion.
Owing to the rare presentation of syngnathia, treatment protocols do not exist. As such, case reports may guide the craniofacial
surgeon for optimal management that may minimize perioperative
and long-term complications. Hegab et al5 reported 3 patients of
congenital maxillomandibular fusion, with complications ranging
from recurrence of fusion, decrease in mouth opening and postoperative mortality. Our patient also experienced recurrence of
fusion early after the initial procedure, potentially related to lack of
compliance in physical therapy. We, however, were able to salvage
this with a repeat osteotomy, placement of an acellular dermal
matrix, and holding the jaws of the patient separated with a neonatal
distractor for 4 weeks. These maneuvers allowed for healing without bony contact and were followed by 3 rounds of jaw manipulation under anesthesia, which did not demonstrate repeat bony
fusion. To our knowledge, this is the first case reported to use
an acellular dermal matrix combined with distractor placement to
achieve long-term separation, normal condylar movement and
growth, though Konas et al9 have reported using a distractor
placement alone.
The embryologic basis of fusion has been studied, but a clear
etiology has not yet been identified. Proposed causes, including
environmental insults, medications such as meclizine, persistence of
the buccopharyngeal membrane, and amniotic constriction bands have
all been postulated.10 As the number of reported cases of syngnathia
reported in the literature increases, optimal management with prevention of recurrence may be further elucidated and established.
REFERENCES
1. Daniels JSM. Congenital maxillomandibular fusion: a case report and
review of the literature. J Craniomaxillofac Surg 2004;32:135139
#
2. Tursken Z, Ozakpinar HR, Tellioglu AT. A case of syngnathia, cleft

palate and hypospadias: an isolated case or syndromic syngnathism?
J Craniomaxillofac Surg 2012;40:810
3. Parkins GE, Bomah MO. Congenital maxillomandibular syngnathia:
case report. J Craniomaxillofac Surg 2009;37:276278
4. Tauro DP, Kallappanavar NK, Kiran HY. Congenital zygo-mandibular
fusion (pseudo-syngnatia?) in conjunction with unilateral anopthalmia:
review of terminology and classification. Cleft Palate Craniofac J
2011;49:626629
5. Hegab A, ElMadawy WM, Shawkat WM. Congenital mxillomandibular
fusion: a report of three cases. Int J Oral Maxillofac Surg 2012;41:
12481252
6. Mortazavi SH, Motamedi MK. Congenital bony syngnathia of the jaws.

Indian J Pediatr 2007;74:416418
7. Gil-Da-Silva-Lopes V, Luquetti D. Congenital temporomandibular joint
ankylosis: clinical characterization and natural history of four unrelated
affected individuals. Cleft Palate Craniofac J 2005;42:498694
8. Sarnat B, Robinson I. Experimental changes of the mandible: a serial
roentgenographic study. J Craniofac Surg 2007;18:917925
9. Konac E, Aliyev A, Tuncbilek G. Congenital maxillomandibular
sygnathia: a new management technique using distraction techniques.
J Craniofac Surg 2015;26:e68e70
10. Dawson KL, Gruss JS. Congenital bony syngnathia: a proposed
classification. Cleft Palate Craniofac J 1997;34:141146
e111

A Novel Technique For Short Nose Correction

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Brief Clinical Studies

The Journal of Craniofacial Surgery

The Age-Related Development

Volume 27, Number 1, January 2016

# 2015 Mutaz B. Habal, MD

The Journal of Craniofacial Surgery

Volume 27, Number 1, January 2016

Brief Clinical Studies

Computed Tomography Procedure

FIGURE 2. Estimation of MSV on coronal CT images by superimposing

The Cavalieri Principle Applied to Computed

FIGURE 1. Measurements for ellipsoid formula (maximal width (B), maximal

2015 Mutaz B. Habal, MD

Coefficient of Error for Point Counting

The Journal of Craniofacial Surgery

Brief Clinical Studies

TABLE 1. Mean Values of Maximal Width, Height, and Depth of MS

Estimation of Maxillary Sinus Volume Using

Right MSV (cm3)

Left MSV (cm3)

MSV, maxillary sinus volume; n, case number.

Volume 27, Number 1, January 2016

Comparison of Stereological Method and

Despite the clinical importance of paranasal sinuses, the real

2015 Mutaz B. Habal, MD

The Journal of Craniofacial Surgery

Volume 27, Number 1, January 2016

Brief Clinical Studies

TABLE 3. Bilateral MSV Results in Both Sex Using Ellipsoid Formula

Left MSV (cm3)

MSV, maxillary sinus volume.

There are many studies evaluating the volume of the paranasal

2015 Mutaz B. Habal, MD

Left MSV ICC

Brief Clinical Studies

The Journal of Craniofacial Surgery

Volume 27, Number 1, January 2016

TABLE 5. Mean Volumes of Bilateral MSs Calculated by Using Stereological

FIGURE 4. The comparison of volume results determined on 3 plans calculated

Right MSV (cm3)

Left MSV (cm3)

FIGURE 5. The comparison of volume results determined on 3 plans calculated

MSV, maxillary sinus volume.

2015 Mutaz B. Habal, MD

The Journal of Craniofacial Surgery

Volume 27, Number 1, January 2016

Brief Clinical Studies

TABLE 6. Bilateral MSV Results in Both Sex Using Stereological Method

Right MSV (cm3)

Left MSV (cm3)

Right MSV (cm3)

Left MSV (cm3)

MSV, maxillary sinus volume.

evaluating the relationship between the stereological method and

ICC, interclass correlation coefficient; Stm, stereological method.

2015 Mutaz B. Habal, MD

FIGURE 7. A BlandAltman analysis of the right MSV as measured by ellipsoid

FIGURE 8. A BlandAltman analysis of the left MSV as measured by ellipsoid

The Journal of Craniofacial Surgery

Brief Clinical Studies

Volume 27, Number 1, January 2016

24. Roberts N, Cruz-Orive LM, Reid NM, et al. Unbiased estimation of

A Novel Technique for Short