You are on page 1of 4

G Model

PEDOT-7560; No. of Pages 4

International Journal of Pediatric Otorhinolaryngology xxx (2015) xxx–xxx

Contents lists available at ScienceDirect

International Journal of Pediatric Otorhinolaryngology


journal homepage: www.elsevier.com/locate/ijporl

Acoustic analysis of voice in children with cleft palate and


velopharyngeal insufficiency
Rocio Villafuerte-Gonzalez a, Victor M. Valadez-Jimenez a,
Xochiquetzal Hernandez-Lopez a, Pablo Antonio Ysunza b,*
a
Department of Speech & Language Pathology, Instituto Nacional de Rehabilitacion, Mexico City, Mexico
b
Ian Jackson Craniofacial and Cleft Palate Clinic, Neuroscience Program, Beaumont Health, Royal Oak, MI, United States

A R T I C L E I N F O A B S T R A C T

Article history: Background: Acoustic analysis of voice can provide instrumental data concerning vocal abnormalities.
Received 2 March 2015 These findings can be used for monitoring clinical course in cases of voice disorders. Cleft palate severely
Received in revised form 17 April 2015 affects the structure of the vocal tract. Hence, voice quality can also be also affected.
Accepted 18 April 2015
Objective: To study whether the main acoustic parameters of voice, including fundamental frequency,
Available online xxx
shimmer and jitter are significantly different in patients with a repaired cleft palate, as compared with
normal children without speech, language and voice disorders.
Keywords:
Materials and methods: Fourteen patients with repaired unilateral cleft lip and palate and persistent or
Cleft palate
Speech
residual velopharyngeal insufficiency (VPI) were studied. A control group was assembled with healthy
Voice volunteer subjects matched by age and gender. Hypernasality and nasal emission were perceptually
Acoustics assessed in patients with VPI. Size of the gap as assessed by videonasopharyngoscopy was classified in
patients with VPI. Acoustic analysis of voice including Fundamental frequency (F0), shimmer and jitter
were compared between patients with VPI and control subjects.
Results: F0 was significantly higher in male patients as compared with male controls. Shimmer was
significantly higher in patients with VPI regardless of gender. Moreover, patients with moderate VPI
showed a significantly higher shimmer perturbation, regardless of gender.
Conclusion: Although future research regarding voice disorders in patients with VPI is needed, at the
present time it seems reasonable to include strategies for voice therapy in the speech and language
pathology intervention plan for patients with VPI.
ß 2015 Elsevier Ireland Ltd. All rights reserved.

1. Introduction Acoustic analysis includes measurement of several parameters.


The most important for clinical purposes are Fundamental
Perceptual assessment of voice is voice is fundamental for the Frequency (F0), Jitter and Shimmer. FO is determined by the
evaluation of patients with voice disorders. Digital acoustic frequency at which the vocal cords vibrate during voice production
analysis of voice provides instrumental data complementing and it is measured in Hz. Normal values of F0 for children have
perceptual assessment. Digital acoustic analysis increases the been previously reported [2]. F0 in normal female children ranges
knowledge concerning specific voice characteristics and it is from 268 to 295 Hz. A mean F0 of 247 Hz in children under 12 years
helpful for understanding the mechanisms of voice production. of age has also been reported, without significant differences when
Moreover, acoustic data are useful for following the patient’s boys and girls were compared [3].
clinical course, documenting clinical improvement and providing During consistent vocal cord vibration, F0 demonstrates a slight
visual biofeedback which can be used during the voice intervention variation and also a variation of cycle-to-cycle amplitude, these
[1]. phenomena are known as frequency perturbation or jitter and
amplitude perturbation or shimmer. These variations or changes
are a consequence of the mass and tension differences which
characterize the vocal cords. Jitter and shimmer values can be
* Corresponding author. Tel.: +1 248 551 2100; fax: +1 248 551 4692. expressed in a percentage. There are normative data for these
E-mail address: antonio.ysunza@beaumont.edu (P.A. Ysunza). percentages. Jitter percentage should be below 1% whereas

http://dx.doi.org/10.1016/j.ijporl.2015.04.030
0165-5876/ß 2015 Elsevier Ireland Ltd. All rights reserved.

Please cite this article in press as: R. Villafuerte-Gonzalez, et al., Acoustic analysis of voice in children with cleft palate and
velopharyngeal insufficiency, Int. J. Pediatr. Otorhinolaryngol. (2015), http://dx.doi.org/10.1016/j.ijporl.2015.04.030
G Model
PEDOT-7560; No. of Pages 4

2 R. Villafuerte-Gonzalez et al. / International Journal of Pediatric Otorhinolaryngology xxx (2015) xxx–xxx

shimmer percentage should be below 10%. It should be considered Patients present with compensatory articulation errors including
that shimmer and jitter are dependent of F0. Thus, in cases of glottal stops were excluded from the study group. Patients with
abnormal voices with aperiodicity which precludes F0 measure- neurological disorders or intellectual – cognitive deficits were
ment, shimmer and jitter are not useful [1]. excluded. Patients with severe nasal emission causing nasal
Systematic changes of F0 can occur as a consequence of stress or rustle which was being picked up during the recordings procedures
intonation, which are inherent to speech. These changes affect the were also excluded.
acoustic assessment of F0. Thus, the clinical evaluation of F0, During the recruiting period, a total of 14 patients met the
shimmer and jitter is usually performed during the production of criteria for being included in the study. Eleven patients were males
a sustained vowel sound. The most acoustically consistent vowel and 3 patients were females. The age of the patients ranged from
sound is the vowel/a/ [4,5]. 7 to 9 years of age with a median of 8 years. The study protocol
Vocal qualities are the result of individual anatomical and was carefully explained to the parents and/or legal guardians and
physiological characteristics which are strongly influenced by age, all of them signed an informed consent form.
gender and physical structure [6]. Therefore, for an adequate vocal
production it is necessary that all the components of the vocal 2.2. Controls
tract, from the glottis to the lips and including the resonance
chambers of the nasal cavities and sinuses be structurally and A control group was assembled, including 14 subjects matched
functionally intact. by age and gender. These subjects were recruited at a nearby
The most common craniofacial anomaly affecting resonance elementary school. All these subjects voluntarily participated in
and speech is a palatal cleft. According to Kernahan and Stark [7], the study. The study protocol was carefully explained to the
the most common cleft is the total cleft of the primary and parents and/or legal guardians and all of them signed an informed
secondary palate. Patients with a cleft of the secondary palate consent form. All of these patients were examined in order to verify
show abnormal nasal resonance and nasal emission, which are a that there were no palatal anomalies (including submucous cleft
consequence of the defective or incomplete seal of the velophar- palate), no voice disorders, no compensatory articulation errors
yngeal sphincter during speech. This anomaly is known as and no perceptual signs of VPI (normal nasal resonance during
velopharyngeal insufficiency (VPI) and significantly affects speech speech).
intelligibility [8]. Besides hypernasality and nasal emission, some
patients with cleft palate also develop compensatory behaviors 2.3. Procedure
known as compensatory articulation errors. These articulation
disorders affect intelligibility even more severely than VPI [9]. In patients with repaired cleft palate and persistent VPI,
Some papers have focused on the acoustic characteristics of hypernasality and nasal emission were perceptually classified as
voice in patients with cleft palate. Frequency instability, high previously reported [13,14]. Two experienced physicians certified
intensity, abnormal formant distribution and abnormal spectro- by the Board of Speech and Language Pathology of Mexico (first
grams have been reported [10–12]. In 1989, Zajac and Linvile [11] and second authors of this paper) were in charge of conducting the
studied voice perturbations in patients with VPI. They performed examination. The examinations were performed independently.
acoustic analyses and found a significant relationship between A Cohen’s Kappa concordance index of 85 was found between both
abnormally high jitter values and hypernasality. examiners. Regarding nasal emission a Cohen’s Kappa index of
The purpose of this paper is to study acoustic parameters of 92 was found. Whenever there was disagreement, each case was
voice including F0, shimmer and jitter in children with persistent discussed until a consensus had been reached. Hypernasality was
or residual VPI after surgical repair of a cleft palate. The acoustic detected in all patients. Only 4 patients presented with nasal
parameters of these children were compared with a group of emission across plosive and fricative phonemes. It should be
normal controls matched by age and gender, without any voice or pointed out that none of these patients showed nasal rustle that
speech disorder. could be picked up by the microphone during the recording
procedures. Eight patients presented with mild hypernasality. Six
2. Materials and methods patients presented with moderate hypernasality.
All patients with repaired cleft palate and persistent VPI
A comparative, cross-sectional study was carried out at the underwent a flexible nasoendoscopy following a protocol which
Department of Speech and Language Pathology of the National has been previously reported [15]. Velopharyngeal movements
Institute of Rehabilitation in Mexico City from January 2012 to during speech including velar and lateral pharyngeal motion, as
December 2013. The protocol was approved by the Internal Review well as presence of Passavant’s ridge were classified as ratios,
Board of the Institute. considering the position at rest as 0 and full contact or closure as
1.0. The size of the gap during speech was estimated as a ratio
2.1. Patients considering total velopharyngeal space at rest as 1.0. Eight patients
demonstrated only bubbling during speech without a discernible
Patients who met the following criteria were recruited for the gap. These cases were conventionally considered as a 10% gap and
study: (a) surgically corrected non-syndromic unilateral total cleft a mild VPI. Six patients demonstrated gaps with a size of 20%, that
lip and palate (UCLP) with persistent or residual VPI as is, a moderate VPI. It should be pointed out that size of the gap as
demonstrated by perceptual analysis of voice and videonasophar- assessed by videonasopharyngoscopy is merely an estimate and
yngoscopy. The patients should present with UCLP as an isolated not hard data. In other words, inter-observer reliability is not
malformation, not associated with a sequence or syndrome; (b) statistically significant. This is why the gaps are conventionally
normal hearing as demonstrated by a behavioral pure – tone assessed in 10-point intervals (10–20–30%, and so on). It should be
audiometry; (c) 6–10 years of age at the time to be recruited for pointed out that the endoscope was passed further into the vocal
the study. This age group was selected in order to assure tract in order to assess the vocal folds. All patients showed normal
appropriate compliance during the behavioral audiometry and mobility of both vocal folds and no masses were identified in any
the acoustic assessment, as well as to prevent voice changes as of the cases. No stroboscopy was performed.
a consequence of puberty. Patients with palatal fistula or with a Acoustic assessments were performed according to the protocol
history of laryngeal pathology or hoarseness were excluded. that was previously reported [16,17]. The patients and subjects

Please cite this article in press as: R. Villafuerte-Gonzalez, et al., Acoustic analysis of voice in children with cleft palate and
velopharyngeal insufficiency, Int. J. Pediatr. Otorhinolaryngol. (2015), http://dx.doi.org/10.1016/j.ijporl.2015.04.030
G Model
PEDOT-7560; No. of Pages 4

R. Villafuerte-Gonzalez et al. / International Journal of Pediatric Otorhinolaryngology xxx (2015) xxx–xxx 3

Table 1
Distribution of acoustic parameters.

Variable Patients Control subjects P; 95% CI Patients Control subjects P; 95% CI

Males Females

F0 270.4 Hz SD  7.01 257.9 Hz SD  6.57 0.000 259.1 Hz SD  35.3 240.2 Hz SD  2.1 0.407
Shimmer 5.2 SD  0.86 0.78 SD  0.21 0.000 7.7 SE  3.5 0.6 SD  0.04 0.024
Jitter 1.24 SD  0.26 0.23 SD  0.09 0.000 1.2 SD  1.8 1.01 SD  0.07 0.826

Table displays mean values and standard deviations (SD) of F0, shimmer and jitter of the groups of patients and control subjects. Patients and subjects were divided by gender
for comparison purposes. Probability values (P) are also included. A 95% confidence interval was selected for considering P values as statistically significant.

were seated in a straight position on a clinical chair. They were 4. Discussion


asked to produce a sustained vowel sound (/a/) for at least 3 s at an
intensity that was considered as comfortable by the patients and From the results of this study it seems that patients present
subjects. A Perfect Choice microphone PC-110279 LO2311027 was with VPI show vocal abnormalities which were expressed by
used at a distance of 10 cm from the lips and an angle of 30 degrees differences in F0 and perturbations of voice frequency (jitter) and
toward the patient. F0, jitter and shimmer were determined. amplitude (shimmer).
It should be considered that although the group of patients was
2.4. Statistical analysis homogeneous because of the relatively strict inclusion and
exclusion criteria, the number of cases studied herein is reduced,
Conventional central tendency measures, including mean and which prevents drawing definite conclusions.
standard deviation were used for analyzing F0, shimmer and jitter. The acoustic abnormalities found in patients present with VPI
Acoustic parameters were compared between groups (active – could be explained by the presence of VPI during speech
patients and control – subjects) through a non-paired two tailed development. VPI occurs most commonly in individuals with a
student t test. The SPSS Statistics 20 software was used for history of cleft palate. Although surgeons attempt to achieve as
performing the statistical tests. much velar lengthening and motion at early ages, through different
surgical techniques, 20–40% of patients with cleft palate will
present with persistent or residual VPI after surgical palatal repair
3. Results [18,19]. Thus, if there is a significant VPI during speech
development it seems plausible that several patients will show
F0 was significantly higher in males from the group of patients vocal compensatory behaviors for the lack of intraoral pressure.
as compared with male controls. In contrast, female patients and It should be pointed out that the patients studied for this paper
subjects did not demonstrate a significant difference (see Table 1). did not present with compensatory articulation errors. Therefore,
Shimmer was significantly higher in the group of patients as the vocal compensatory behaviors mentioned herein seem to be
compared with controls, regardless of gender (see Table 1). These independent of compensatory articulation errors such as glottal
data suggest that patients with persistent VPI seem to show higher stops and pharyngeal fricatives.
variability of peak-to-peak amplitude between consecutive Patients with VPI showed acoustic abnormalities which may be
periods. associated with vocal fold adduction and sudden release. Patients
Jitter was significantly higher in males from the group of with vocal fold paresis show similar although more severe acoustic
patients as compared with male controls. Female patients and findings, but the acoustic findings in patients with VPI may be
subjects did not demonstrate a significant difference (see Table 1). indicating a low concentration of acoustic energy at the F0 and
These data suggest that male patients with persistent VPI seem to relative to the overall energy of the signal.
show higher variability in F0 signal periodicity. Patients present with VPI may be attempting to regulate active
When patients present with VPI with nasal emission were vocal tract resistances as compensation. Moreover, increased
compared with patients with VPI without nasal emission, non- glottal resistances during vowel production would decrease air
significant differences in F0, shimmer or jitter were found. flow and hence facilitate the regulation of subglottal pressure
When the patients were stratified according to the severity of which is essential for continuous phonation [20].
VPI (mild to moderate), a significant difference was demonstrated A possible explanation for some of the acoustic abnormalities
between shimmer values of patients with mild VPI (10% gaps) described herein, especially the apparent relationship between
as compared with patients with moderate VPI (20% gaps). Patients severity of VPI and shimmer perturbation, may be that the
with moderate VPI showed a higher shimmer perturbation. This individual is attempting to regulate subglottal pressure and vocal
difference was independent of gender (see Table 2). tract aerodynamics in a condition in which there is insufficient
coupling between the resonant nasal cavities and the rest of the
vocal tract [21], potentially increasing vocal noise.
Table 2 Another interesting finding of this study was gender dependent
Comparison by severity of VPI. F0 difference. Males showed higher F0 as compared with male
Variable VPI P controls. Moreover, jitter was also higher in male patients as
compared with male controls. These data suggest that male
Mild Moderate
patients with persistent VPI seem to show higher variability in F0
F0 263.2 SD  11.9 274.4 SD  19.3 0.204 signal periodicity.
Shimmer 4.7 SD  0.38 7.1 SD  2.27 0.011
Van Lierde et al. [22] described vocal quality characteristics in
Jitter 0.20 SD  0.09 0.79 SD  1.2 0.207
children with cleft palate through a multiparameter approach.
Table displays mean values and standard deviations (SD) of F0, shimmer and jitter They reported that the results of their study supported the
in the group of patients with velopharyngeal insufficiency (VPI). VPI was classified
according to size of the gap and hypernasality (mild to moderate). Probability (P)
hypothesis that vocal quality disorders characterized by a more
values are also included. A 95% confidence interval was selected for considering P negative DSI-value occur in male cleft palate children. These
values as statistically significant. authors also described that decreased vocal quality expressed as

Please cite this article in press as: R. Villafuerte-Gonzalez, et al., Acoustic analysis of voice in children with cleft palate and
velopharyngeal insufficiency, Int. J. Pediatr. Otorhinolaryngol. (2015), http://dx.doi.org/10.1016/j.ijporl.2015.04.030
G Model
PEDOT-7560; No. of Pages 4

4 R. Villafuerte-Gonzalez et al. / International Journal of Pediatric Otorhinolaryngology xxx (2015) xxx–xxx

acoustic abnormalities in male children with cleft palate may be [5] M. Grellet, J.C. Pereira, M. Oliveira Rosa, Standardization of acoustic measures of
the normal voice, Braz. J. Otorhinolaryngol. 68 (2002) 540–544.
caused by a direct laryngeal pathology or may be an indirect [6] S. Maturo, C. Hill, G. Burting, C. Balif, R. Maurer, C. Hartnick, Establishment of
response to VPI, which supports the hypothesis that vocal a normative pediatric acoustic data base, Otolaryngol. Head Neck Surg. 138 (2012)
hyperfunction and abnormal vocal quality in children with VPI 956–961.
[7] D.A. Kernahan, R.B. Stark, A new classification for cleft lip and cleft palate, Plast.
represent the children’s attempts to compensate for the VPI. Reconstr. Surg. 22 (1958) 435–441.
Although age does not affect mean F0, shimmer and jitter are [8] A. Ysunza, M. Pamplona, F. Molina, M. Drucker, J. Felemovicius, E. Ramirez, et al.,
age related. Thus, the rather broad age range of the patients studied Surgery for speech, Int. J. Pediatr. Otorhinolaryngol. 68 (2004) 1499–1505.
[9] M.C. Pamplona, A. Ysunza, S. Morales, Strategies for treating compensatory
in this paper could have affected the acoustic assessments. articulation in patients with cleft palate, Int. J. Biomed. Sci. 10 (2014) 43–51.
Moreover, in this study, nasal resonance and nasal emission were [10] R. Kataoka, D.W. Warren, D.J. Zajac, R. Mayo, R.W. Lutz, The relationship between
assessed perceptually using a previously published classification spectral characteristics and perceived hypernasality in children, J. Acoust. Soc.
Am. 16 (2001) 205–210.
which has not been entirely validated concerning its reliability. It
[11] D.J. Zajac, R.N. Linville, Voice perturbations of children with perceived nasality
will be necessary to perform future studies using instrumental and hoarseness, Cleft Palate J. 26 (1989) 226–231.
assessments including nasometry and aerodynamics measure- [12] J. Chenghui, T.L. Whitehill, B. McPherson, M. Lawrence, Spectral features and
ments for assessing nasal resonance and emission. perceptual judgment of place of affricate in Putonghua-speaking pre-adolescents
with normal and cleft palate, Int. J. Pediatr. Otorhinolaryngol. 79 (2015) 179–185.
[13] G. Henningsson, D.P. Kuehn, D. Sell, T. Sweeney, J. Trost-Cardamone, T. Whitehill,
5. Conclusion et al., Universal parameters for reporting speech outcomes in individuals with
cleft palate, Cleft Palate Craniofac. J. 45 (2008) 1–17.
[14] A. Ysunza, A. Pamplona, S. Morales, Velopharyngeal valving during speech, in
The results of this study suggest that acoustic parameters of patients with velocardiofacial syndrome and patients with non-syndromic clefts
voice are significantly affected in patients with VPI. Jitter seems after surgical and speech pathology management, Int. J. Pediatr. Otorhinolaryn-
gol. 75 (2011) 1255–1259.
to be more affected in male patients and there seems to be a [15] A. Ysunza, R. Merson, Videonasopharyngoscopy of the velopharyngeal sphincter
relationship between severity of VPI and perturbation of shimmer. during speech and swallowing, in: D.M. Grange (Ed.), Endoscopy and Endoscopic
The vocal abnormalities found in this study seem to be associated Procedures: Management, Technologies and Methods of Improvement, Nova,
New York, 2014, , ISBN: 978-1-63117-0805.
with compensatory behaviors related with VPI and they are [16] V. Valadez, A. Ysunza, E. Ocharan-Hernandez, N. Garrido-Bustamante, A. Sanchez-
independent of compensatory articulation errors such as glottal Valerio, M.C. Pamplona, Voice parameters and videonasolaryngoscopy in children
stops. Patients present with VPI may be at risk of developing vocal with vocal nodules. A longitudinal study before and after voice therapy, Int. J.
Pediatr. Otorhinolaryngol. 76 (2012) 1361–1365.
pathology. Future research regarding vocal disorders in patients
[17] A. Garcı́a Pérez, X. Hernández-López, V.M. Valadez, A. Minor-Martı́nez, A. Ysunza,
with VPI is needed, including collection of vocal tract resonance Synchronous electrical stimulation of laryngeal muscles: an alternative for en-
measures (spectrograms and cepstral peak prominence measure- hancing recovery of unilateral recurrent laryngeal nerve paralysis, J. Voice 28
ments). However, at the present time, it seems reasonable to (2014) 524e1–524e7.
[18] A. Ysunza, K. Shaheen, D.J. Aughton, M.A. Micale, R. Merson, K. Rutkowski,
include strategies for voice therapy in the speech & language Velopharyngeal insufficiency, submucous cleft palate and a phonological disorder
pathology intervention plan for patients with VPI. as the associated clinical features leading to the diagnosis of Jacobsen syndrome.
Case report and review of the literature, Int. J. Pediatr. Otorhinolaryngol. 77
(2013) 1601–1605.
References [19] J. Nyberg, P. Peterson, A. Lohmander, Speech outcomes at age 5 and 10 years in
unilateral cleft lip and palate after one-stage palatal repair with minimal incision
[1] K.C. Toran, B.K. Lal, Objective analysis of voice in normal young adults, Kathmandu technique – a longitudinal perspective, Int. J. Pediatr. Otorhinolaryngol. 78 (2014)
Univ. Med. J. 7 (2009) 374–377. 1662–1670.
[2] L. Wanderley Lopes, S.L. Do Nascimento Cunha Costa, W.C. De Almeida Costa, S.E. [20] Z. Yang, J. Fan, J. Tian, L. Liu, C. Gan, W. Chen, et al., Cepstral analysis in children
Nobrega Correia, V.J. Dias Vieira, Acoustic assessment of the voices of children with velopharyngeal insufficiency after cleft palate surgery, J. Voice 28 (2014)
using nonlinear analysis: proposal for assessment and vocal monitoring, J. Voice 789–792.
28 (2014) 565–573. [21] S. Ha, H. Sim, M. Zhi, D.P. Kuehn, An acoustic study of the temporal characteristics
[3] M. Guzman, D. Munoz, M. Vivero, N. Marin, M. Ramirez, M.T. Rivera, et al., Acoustic of nasalization in children with and without cleft palate, Cleft Palate Craniofac. J.
markers to differentiate gender in prepubescent children’s speaking and singing 41 (2004) 535–543.
voice, Int. J. Pediatr. Otorhinolaryngol. 78 (2014) 1592–1598. [22] K.M. Van Lierde, S. Claeys, M. De Bodt, V. Van Cauwenberge, Vocal quality
[4] E.T. Doherty, Evaluation of selected acoustic parameters for use in speaker characteristics in children with cleft palate: a multiparameter approach, J. Voice
identification, J. Acoust. Soc. Am. 58 (S107) (1975) 1121. (2004) 354–362.

Please cite this article in press as: R. Villafuerte-Gonzalez, et al., Acoustic analysis of voice in children with cleft palate and
velopharyngeal insufficiency, Int. J. Pediatr. Otorhinolaryngol. (2015), http://dx.doi.org/10.1016/j.ijporl.2015.04.030

You might also like