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Auris Nasus Larynx 39 (2012) 163168

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A cupulolith repositioning maneuver in the treatment


of horizontal canal cupulolithiasis
Sung Huhn Kim a, Sung-Woo Jo b, Woon-Kyo Chung c, Hyung Kwon Byeon a, Won-Sang Lee a,*
a

Department of Otorhinolaryngology, Yonsei University College of Medicine, 134 Shinchon-dong, Seodaemun-gu, 120-752 Seoul, Republic of Korea
b
Ilsan Yonsei ENT Clinic, Goyang-si, Gyeonggi-do, Republic of Korea
c
Shinchon Yonsei ENT Clinic, Seoul, Republic of Korea
Received 20 January 2011; accepted 26 March 2011
Available online 1 June 2011

Abstract
Objective: We evaluated outcomes and their significance of a new treatment method for horizontal canal cupulolithiasis that could be applied
regardless of the side of the cupula where otoliths are attached.
Methods: Consecutive 78 patients who showed persistent apogeotropic horizontal canal positional vertigo (horizontal canal cupulolithiasis)
were enrolled, and they were treated with the new cupulolith repositioning maneuver.
Results: Horizontal semicircular canal cupulolithiasis was alleviated in 97.4% of patients, after an average of 2.1 repetitions of the maneuver.
Otoliths were suspected to be attached to the canal side of the cupula in 30 cases and the utricular side in 44 cases.
Conclusion: The cupulolith repositioning maneuver is an effective method for treating horizontal canal cupulolithiasis. It may also provide an
insight into the side of the cupula where otoliths are attached.
# 2011 Published by Elsevier Ireland Ltd.
Keywords: Benign paroxysmal positional vertigo; Horizontal semicircular canal; Cupulolithiasis; Treatment

1. Introduction
Positional vertigo caused by the effects of free-floating
(canalolithiasis) or cupula-attached otoliths (cupulolithiasis)
on the flow of endolymph in the semicircular canals is one of
the most common peripheral vestibular disorders. The
posterior canal is the most commonly involved site, and
benign paroxysmal positional vertigo (BPPV) of posterior
canal typically has a good resolution rate (9095%) after
patients undergo a repositioning maneuver [1,2]. Otoliths in
the horizontal canal can also cause positional vertigo which
occurs less frequently than posterior canal BPPV (1020%),
and the resolution rate after conventional repositioning
maneuvers for horizontal canal positional vertigo is poorer
(7080%) than the posterior canal type [35]. There are
* Corresponding author. Tel.: +82 2 2228 3606; fax: +82 2 393 0580.
E-mail addresses: wsleemd@yumc.yonsei.ac.kr, wsleemd@yuhs.ac
(W.-S. Lee).
0385-8146/$ see front matter # 2011 Published by Elsevier Ireland Ltd.
doi:10.1016/j.anl.2011.03.008

several variants in the horizontal canal positional vertigo,


and it can be classified into three forms according to the
characteristics of nystagmus which can be appeared on the
head roll test [6]. First, it can be positional vertigo showing
transient geotropic nystagmus, and this is thought to be
caused by canalolithiasis in the posterior part of the
horizontal canal. Another form is positional vertigo which
initially shows apogeotropic nystagmus and then changes to
a geotropic form during repetitive head roll test or during
repositioning maneuvers. This can be explained by
canalolithiasis in the anterior part or the horizontal canal.
Finally, positional vertigo could show persistent apogeotropic nystagmus during head roll test and no changes in the
direction of nystagmus even after repetitive head roll test or
repositioning maneuvers. It can be explained by the
cupulolithiasis of the horizontal canal.
Although the precise locations of otolith attachment have
not been proven in horizontal canal cupulolithiasis, otoliths
can theoretically be attached to the cupula of the ampulla on

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S.H. Kim et al. / Auris Nasus Larynx 39 (2012) 163168

either the canal side or the utricular side [4,7]. Both types of
cupulolithiasis may show the same kind of apogeotropic
horizontal nystagmus during a head roll test. However, their
responses to repositioning maneuvers can be different, so
each canal-sided and utricular-sided cupulolithiasis should
be treated with repositioning maneuvers from different
directions. At present, it is not easy to identify whether
otoliths are attached to the utricular side or the canal side of
the cupula, and there is no standardized treatment for
cupulolithiasis. Therefore, we designed a new treatment
method for persistent apogeotropic horizontal canal
positional vertigo (horizontal canal cupulolithiasis) which
can be applied regardless of the side of cupula where
the otoliths are attached. We also have performed this
method for several years. In this study, we report a new
repositioning maneuver and discuss its treatment outcomes
and significance.

2. Patients and methods


2.1. Cupulolith repositioning maneuver for
cupulolithiasis of the horizontal semicircular canal
We produced a diagram of the horizontal semicircular
canal, ampulla, and utricle based on the picture of the human
horizontal semicircular canal taken by Curthoys et al. [8] and
used it to design the repositioning maneuver for persistent
apogeotropic horizontal canal positional vertigo (cupulolith
repositioning maneuver, CuRM) (Fig. 1). Begin with the
patient in the supine position. In this position, the patient will
not show nystagmus or show weak horizontal nystagmus to
the lesion side as Bisdorff and Debatisse described [9].
We observed weak horizontal nystagmus in 15 patients
(19.2%). Turn the patients head 1358 to the lesion side (1st
position). While observing the weak apogeotropic horizontal

Fig. 1. Schematic drawing of the cupulolith repositioning maneuver in a case with right horizontal canal cupulolithiasis. Dash-lined circles in the ampullary
area indicate the exit of the anterior semicircular canal to the utricle. Otoliths are attached to the canal side and the utricular side of the cupula. The rectangle in
the 1st and 4th positions indicates the application of a hand-held vibrator to the suprameatal triangle for 20 s.

S.H. Kim et al. / Auris Nasus Larynx 39 (2012) 163168

165

Fig. 2. Illustration of an incorrect cupulolith repositioning maneuver. In the lateral decubitus position with the head rotated 908 to the affected side, vibrating the
suprameatal area can cause otoliths which attached to the canal side of the cupula become to fall toward the ampulla. If we proceed with head rotation to the
opposite side from this position, the otoliths will remain in the ampulla. Dash-lined circles in the ampullary area indicate the exit of the anterior semicircular
canal to the utricle. The rectangle in the 1st position indicates the application of a hand-held vibrator to the suprameatal triangle for 20 s.

nystagmus, oscillate the suprameatal triangle in the posterior


superior area of the lesion side auricle with a 60 Hz hand-held
vibrator (UM-30, Maxtar, Gimpo, Korea) for 30 s to detach
otoliths from the cupula. The patients head is turned 1358 in
the 1st position because the detached otoliths may remain in
the ampulla of the canal after oscillation if the patients head is
turned 908 to the lesion side (Fig. 2). Nystagmus and vertigo
will disappear after oscillation if otoliths were attached only to
the canal side of the cupula. If otoliths are attached to the
utricular side of the cupula, nystagmus will persist as long as
the patient remains in this position.
Next, turn the patients head 458 to the healthy side (2nd
position, lateral decubitus to lesion side). Transient weak
apogeotropic nystagmus will show if the otoliths are attached
only to the canal side of the cupula, because the detached
otoliths function as canalolithiasis which induces ampullofugal flow of the endolymph. However, if otoliths attached to the
utricular side of the cupula, apogeotropic nystagmus persists.
Next, turn the patients head 908 to the healthy side (3rd
position, supine position). Transient horizontal nystagmus
beating to the healthy side will appear because detached

otoliths function as canalolithiasis in cases where otoliths


were attached only to the canal side of the cupula, as
described in the 2nd position.
For the 4th position, turn the patients head 908 to the
healthy side (lateral decubitus to the healthy side). If a strong
apogeotropic nystagmus is observed, otoliths are likely to be
attached to the utricular side of the cupula. Then, oscillation
will be required once again. However, if only transient
geotropic nystagmus is observed, the detached otoliths have
moved toward the utricle, and oscillation is not necessary.
For the 5th position, rotate the patients head 908 in the
same direction (5th position, prone position), and slowly
bring the patient to a sitting position without neck extension.
Each position in the maneuver should be maintained for
3 min to allow all the detached otoliths to be completely
repositioned. The night after the repositioning maneuver,
patients should sleep in the lateral decubitus position on the
healthy side to prevent the otoliths from re-entering the
semicircular canal and to stabilize the repositioned otoliths
in the utricle. The changes in nystagmus according to the
position of CuRM are summarized in Table 1.

Table 1
Changes of nystagmus according to the otolith-attached side of the cupula during cupulolith repositioning maneuver (CuRM).
Patients position during CuRM

1st position
2nd position
3rd position
4th position
5th position
*

Otolith-attached side of the cupula.

Character and direction of nystagmus


Canal side*

Utricular side *

Persistent apogeotropic, disappear after oscillation


Transient apogeotropic
Transient nystagmus beating to healthy side
Transient geotropic
Transient nystagmus beating to healthy side

Persistent apogeotropic regardless of oscillation


Persistent apogeotropic
No nystagmus
Persistent apogeotropic, disappear after oscillation
Transient nystagmus beating to lesion side

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S.H. Kim et al. / Auris Nasus Larynx 39 (2012) 163168

2.2. Patient selection and evaluation of treatment


outcomes
The newly designed CuRM was performed on 78
consecutive patients who were diagnosed with horizontal
canal cupulolithiasis at Severance Hospital between January
1998 and December 2008. This study was approved by the
institutional review board of Yonsei University College of
Medicine, and patients agreed to undergo CuRM. Patients
with horizontal canal cupulolithiasis combined with vertical
canal BPPVand those who were suspicious of central nervous
system disorder on neurologic examinations and laboratory
tests were excluded from this study. To evaluate peripheral
vestibular function, patient histories were thoroughly
reviewed, and all patients underwent pure tone audiometry
and bithermal caloric tests. Of the 78 patients, 35 were male
and 43 were female, and the mean age at onset was 62.5 years
(range: 2787 years). The lesions were on the right side in 40
patients, on the left in 34, and could not be determined by the
head roll test in 4 cases. The mean follow-up period was 29.8
months (range: 1054 months). Brain magnetic resonance
imaging (MRI) with angiography was performed in patients
who showed intractable positional nystagmus and vertigo
even after repeatedly undergoing CuRM to rule out pathologic
conditions of the central nervous system. A diagnosis of
horizontal canal cupulolithiasis was made based on the head
roll test with electronystagmography (Nicolet Nystar Plus,
version 4.33, Warwick, UK) and video nystagmography
(SensoMotoric Instruments, Berlin, Germany). To determine
if the apogeotropic nystagmus is persistent, we observed
patients nystagmus for more than 2 min and performed the
head roll test 3 times. This time, fatigability of nystagmus
should not be noticed. We did not include controls who would
not undergo any repositioning maneuver or sham maneuver
for ethical reasons. The classic method described by Nuti et al.
[6] was applied to determine the affected side (head roll test):
test on the lesion side evoked weak apogeotropic horizontal
nystagmus, while test on the healthy side evoked strong
nystagmus. If there was no significant difference between
nystagmus intensities (difference of peak slow phase velocity
<108/s), we performed CuRM on one side, then repeated the
head roll test. If nystagmus and vertigo subsided, the side at
which CuRM was performed was considered to be the lesion
side. If nystagmus and vertigo persisted, CuRM was
performed on the other side. In these cases, we assumed
that if the symptoms subsided after treatment, either the lesion
was on the side where the second CuRM had been performed
or bilateral. Patients were followed up 2 days after treatment
and underwent positioning tests such as the head roll test and
the Dix-Hallpike test. If no nystagmus or vertigo appeared,
treatment ended. If nystagmus and vertigo reappeared, a
repositioning maneuver for the involved canal was repeated,
and patients were followed up 2 days later with the same
procedure. We adopted Epleys definition of complete
resolution [10] which is no further positional nystagmus or
vertigo for 1 month after the last treatment. For the evaluation

of treatment outcomes/success rate, the number of treatments


required for complete remission of positional nystagmus was
analyzed.
3. Results
In all cases, nystagmus developed without latency upon the
head roll tests and could be continuously observed while the
position was maintained. There was no nystagmus fatigue and
no change in the direction of nystagmus upon repeated tests.
Five cases showed vestibular weakness on the lesion side in
the bithermal caloric test. Two of these patients had suffered
from acute vestibular neuritis, 2 of them had histories of
Menieres disease, and 1 had a history of head trauma. Most
patients showed side differences in nystagmus intensity
during the head roll test; however, 5 patients showed no
significant differences between the intensity of the nystagmus
on each side during the head roll test. In 1 of the 5 patients,
nystagmus subsided after CuRM on one side; in 2 patients, the
nystagmus subsided after CuRM on both sides; and in the
remaining 2 patients, nystagmus persisted until the most
recent follow-up date despite CuRM on both sides.
In 76 (97.4%) of 78 patients, typical nystagmus and vertigo
completely subsided after CuRM. The mean number of
treatments performed on each patient was 2.1  1.7
(mean  SD), and nystagmus disappeared in 48 patients
(61.5%) with single treatment (Table 2). Two patients showed
intractable positional vertigo and nystagmus despite repetition of CuRM as described above, and their brain MRI with
angiography was normal. They had neither unilateral
vestibular weakness nor history of vestibular disease. We
are now carefully following these patients and are considering
canal occlusion surgery for them.
We could infer the side of the cupula to which otoliths were
attached in 74 out of 78 cases by nystagmus changes during
CuRM. Persistent apogeotropic nystagmus disappeared and
transient nystagmus appeared after the 2nd position in 30
patients during the treatment, suggesting that otoliths were
attached only to the canal side of the cupula. Forty-four
patients showed persistent apogeotropic nystagmus until the
4th position, which suggests that otoliths were attached to the
utricular side or both the utricular and canal sides. In 4 cases,
we could not infer the side of cupula where otoliths were
attached; nystagmus and other symptoms disappeared after
Table 2
Number of attempts of cupulolith repositioning maneuver performed on
patients with horizontal canal cupulolithiasis (n = 78).
Number of treatments

Number of patients (%)

1
2
3
4
5
6
Intractable
Mean number of treatments

48 (61.5)
6 (7.7)
6 (7.7)
4 (5.1)
7 (9.0)
5 (6.4)
2 (2.6)
2.1  1.7

S.H. Kim et al. / Auris Nasus Larynx 39 (2012) 163168

CuRM to both sides in 2 cases, and nystagmus and symptoms


persisted even after multiple CuRM in the other 2 cases.
The disease recurred in 8 (10.8%) cases, and the mean
duration from disease control to recurrence was 9.1 months
(range: 126 months). Positional vertigo due to horizontal
semicircular canal cupulolithiasis recurred in 2 cases, and
canalolithiasis of the posterior semicircular canal and
horizontal semicircular canal recurred in 3 cases each. All
recurrent cases were successfully treated with appropriate
repositioning maneuvers for each canal, such as CuRM and
modified Epleys maneuver [1].

4. Discussion
Although various diagnostic evaluation methods and
treatment modalities have been proposed, it is still difficult
and controversial to diagnose and treat horizontal canal
cupulolithiasis. First, otoliths may be attached to either the
utricular or the canal side of the cupula. This has not been
verified histopathologically; however, if this can happen, it is
difficult to determine the side to which otoliths are attached
with the head roll test, which makes the treatment difficult.
Otoliths attached to the canal side of the cupula can be easily
repositioned if a repositioning maneuver is performed to the
lesion side, whereas otoliths on the utricular side remain
attached and induce positional vertigo even after treatment.
Most conventional repositioning maneuvers do not consider
this. From this point of view, our new method is useful in
both identifying the side of the cupula where otoliths are
attached and treating horizontal semicircular canal cupulolithiasis regardless of the side (although CuRM findings
offer indirect evidence of the affected side). In our study, 30
patients were assumed to have cupulolithiasis on the canal
side of the cupula, and 44 patients were assumed to have
cupulolithiasis on the utricular side. Otoliths detached from
the utricle are more likely to enter the ampulla directly from
the utricle than to enter the ampulla through the semicircular
duct. Therefore, we think that the number of utricular-sided
(or both utricular- and canal-sided) cupulolithiasis cases was
larger than canal-side-only cupulolithiasis. This coincides
with the findings of a study by Chiou et al. [11].
Second, the induced apogeotropic nystagmus was
symmetric occasionally, making lateralization difficult. In
such a case, one should consider unilateral vestibular
weakness or cupulolithiasis combined with canalolithiasis or
bilateral cupulolithiasis. If the patient shows unilateral
vestibular weakness on the lesion side, turning the patients
head to the healthy side evokes weaker apogeotropic
nystagmus to the lesion side than nystagmus in the patient
without canal paresis. That is because cupula deviation to
the utriculopetal direction evokes weaker apogeotropic
nystagmus in patients with unilateral vestibular weakness.
Therefore, the intensities of nystagmus can be symmetric. If
canalolithiasis is combined with cupulolithiasis in the same
canal or in the other horizontal canal, it can reduce the

167

intensities of apogeotropic nystagmus by inducing geotropic


nystagmus. However, it is difficult to differentiate these
pathologies with only a head roll test, and CuRM should be
performed on one side first and then on the other side if
nystagmus persists in the head roll test after the first
treatment. This procedure can be used to treat bilateral
cupulolithiasis as well as cupulolithiasis combined with
canalolithiasis on either side because free-floating otoliths
can be moved to the utricle along with the otoliths detached
from the cupula during the maneuver. In our study, 5 patients
(6.4%) showed no differences in the intensity of evoked
nystagmus on both sides. Two of them (2.6%) had unilateral
vestibular weakness. Two of them (2.6%) had a history of
vestibular neuritis, and the other one (1.2%) Menieres
disease. Among the 5 patients, the lesion side could be easily
determined in only 1 case (1.2%), in which persistent
apogeotropic nystagmus subsided in the 1st position of
CuRM in the first trial and turned to transient apogeotropic
nystagmus in the 2nd position, suggesting that the side
where CuRM performed was the lesion side and that otoliths
were attached only to the canal side of the cupula. In the
other 2 cases (2.6%), persistent apogeotropic nystagmus did
not subside after CuRM was performed in one direction.
Instead it subsided after the 4th position of CuRM performed
in the other direction. This suggests that the patients at least
had utricular-sided cupulolithiasis at the side where the
second CuRM was performed. However, bilateral cupulolithiasis cannot be ruled out because CuRM was performed
in both directions. In another 2 cases (2.6%), nystagmus was
intractable even after repeated CuRM trials in both
directions until the most recent follow-up. OReilly et al.
suggested that patients with BPPV refractory to repositioning maneuvers are more likely to have other vestibular or
CNS pathologies [12]. However, they showed no abnormalities on vestibular function test, neurological examinations, or brain MRI with angiography. It is conceivable that
innate narrowing and/or an otoconial jam and/or a fold of the
semicircular canal could cause intractable positional
nystagmus and vertigo, as described in several studies
[1315]. We are now carefully following these patients and
are considering canal occlusion surgery for them.
The fact that persistent nystagmus became transient
apogeotropic nystagmus during CuRM might indicate that
the patient had canalolithiasis located in the ampulla or
anterior part of the horizontal canal, not cupulolithiasis.
However, it should be noted that nystagmus had persisted
and did not change to transient geotropic nystagmus after
repeated head roll test. Therefore, we regarded it as a
cupulolithiasis rather than canalolithiasis.
We used oscillation to detach otoliths from the cupula.
There have been several reports and controversies about the
efficacy of oscillation during repositioning maneuver. While
Li emphasized the importance of oscillation during the
repositioning maneuver [16], but others reported that
oscillation made no difference [1719]. Patients enrolled in
these studies had posterior canal BPPV, and there have been

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S.H. Kim et al. / Auris Nasus Larynx 39 (2012) 163168

no report about the efficacy of oscillation during the treatment


of persistent apogeotropic horizontal canal positional vertigo.
We believe that oscillation is effective in detaching otoliths
from the cupula, as seen in the treatment outcome of CuRM.
However, to validate the efficacy of oscillation for persistent
apogeotropic horizontal canal positional vertigo, a strict
controlled study should be performed.
There have been several reports about the therapeutic
effect of reposition maneuvers on cupulolithiasis of the
horizontal canal. Steenerson et al. used Hains maneuver to
treat horizontal canal BPPV and reported that the maneuver
was successful in all 6 cupulolithiasis patients with repetition
of 3.1 times [20]. Chiou et al. used a forced prolonged position
which is lying on the side of weaker nystagmus, for 40
patients, and vertigo and nystagmus were relieved in all
patients [11]. White et al. used several maneuvers such as
Lempert maneuver, Gufonis maneuver, Vannucchi-Asprella
maneuver, and Brandt Daroff exercise for 10 patients because
none of the maneuvers is universally effective for horizontal
canal cupulolithiasis [21]. They found that only 50% of
patients were relieved from vertigo and nystagmus after these
maneuvers. Casani et al. used a modified Semont maneuver to
9 patients and 77.7% of them showed response [22].
According to White et al. and Casani et al., horizontal canal
cupulolithiasis showed poorer response to reposition maneuvers than canalolithiasis. Steenerson et al. showed good
result with Hains maneuver. However, they had a small
number of patients with horizontal canal cupulolithiasis, and
the repetition number of treatment was higher than that in our
study. Chiou et al. also showed good results, but his method
requires patients lie in one position for more than 12 h, which
is uncomfortable to some patients. CuRM relieved vertigo
with an average of 2.1 times of repetition in most patients in
less than 20 min, which will not distress patients much. Thus,
CuRM is an effective method for treating both utricular- and
canal-sided horizontal canal cupulolithiasis at the same time.
It can be easily performed with minimal patient discomfort
because it can be completed quickly at bedside, and patients
can return to their daily work immediately after treatment if
they do not extend their necks or lie down during working
hours. It showed a high success rate (97%) and also provided
an insight that otoliths can be attached to either the canal or the
utricular side of the cupula.
Conflict of interests
None.
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