Keywords balance diseases - cupulolithiasis - head-shaking maneuver - HSC-BPPV - supine roll
test
Introduction
Benign paroxysmal positional vertigo (BPPV) is a mechanical disorder of the vestibular
membranous labyrinth. The main symptom is sudden vertigo provoked by changes in the
position of the head relative to gravity: sitting upright from supine, lying down,
and rolling over in bed, looking up, or stooping forward.[1 ]
[2 ] Symptoms may last for days, weeks, months, or years, or could be recurrent.[3 ] BPPV is commonly due to free-floating otoconial debris entering one or more of three
semicircular canals from the utricle, which is called canalolithiasis.[4 ]
[5 ] Uncommonly, it is due to cupulolithiasis, in which otoconial debris adheres to the
gelatinous cupula.[3 ]
[6 ]
[7 ] Such pathologies result in cupular deflection when the head moves to a certain position,
which is secondary to the otoconial debris overcoming the hydrodynamic resistance
of the endolymph in canalolithiasis and the cupula becoming heavier in cupulolithiasis.[8 ] In either case, it results in asymmetrical stimulation of the vestibular labyrinth
in situations when the head moves relative to gravity, which explains the symptom
of positionally triggered vertigo.[9 ]
[Table 1 ] shows that 1.94 to 38% of all BPPV patients diagnosed at any specialty clinic have
horizontal semicircular canal BPPV (HSC-BPPV).[10 ]
[11 ]
[12 ]
[13 ]
[14 ]
[15 ]
[16 ]
[17 ]
[18 ]
[19 ]
[20 ]
[21 ]
[22 ] The supine roll test (SRT) elicits horizontal positional nystagmus (PN) in HSC-BPPV.[23 ] Apogeotropic PN is ascribed to either cupulolithiasis or canalolithiasis in the
ampullary short anterior arm,[24 ]
[25 ]
[26 ] with cupulolithiasis being categorized as either Cup-C or Cup-U depending on the
side to which otoconial mass is adherent.[6 ]
[7 ]
[27 ] Geotropic PN is seen with nonampullary long posterior arm horizontal semicircular
canalolithiasis.[28 ] Both will present bilaterally during SRT. The duration of the PN is up to 1 minute
in the long posterior arm horizontal semicircular canalolithiasis and more than 1
minute in the cupulolithiasis. The current diagnostic criteria for horizontal semicircular
cupulolithiasis (HSC-BPPV-cu ) require that an apogeotropic horizontal PN elicited during SRT lasts more than a
minute and remains unchanged during repetitive testing.[29 ]
[30 ]
Table 1
Pooled data from 13 studies with frequency of different variants of BPPV attending
a specialty clinic
Authors
No. of patients
PSC-BPPV
HSC-BPPV
ASC-BPPV
Multiple canals
Abbreviations: ASC-BPPV, anterior semicircular canal benign paroxysmal positional
vertigo; HSC-BPPV, horizontal semicircular canal benign paroxysmal positional vertigo;
PSC-BPPV, posterior semicircular canal benign paroxysmal positional vertigo.
De la Meilleure et al,[10 ] 1996
287
78.05%
16.38%
–
5.57%
Honrubia et al,[11 ] 1999
292
85.62%
5.14%
1.37%
7.87%
Macias et al,[12 ] 2000
259
93.02%
1.94%
–
5.04%
Korres et al,[13 ] 2002
122
90.16%
8.2%
1.64%
–
Sakaida et al,[14 ] 2003
50
56%
38%
–
6%
Imai et al,[15 ] 2005
108
64.82%
33.33%
–
1.85%
Nakayama and Epley,[16 ] 2005
833
66.39%
10.08%
2.28%
21.25%
Cakir et al,[17 ] 2006
169
85.21%
11.83%
1.18%
1.78%
Moon et al,[18 ] 2006
1,692
60.9%
31.9%
2.2%
5.0%
Jackson et al,[19 ] 2007
260
66.9%
11.9%
21.2%
–
Chung et al,[20 ] 2009
589
61.8%
35.3%
2.9%
–
Vlastarakos et al,[21 ] 2019
96
90.62%
8.33%
1.04%
–
Chua et al,[22 ] 2020
1,542
92.2%
3.7%
–
4.08%
Cupulolithiasis was first reported in 1969 as a granular basophilic mass attached
to the cupula of the posterior semicircular canal in the temporal bone histological
sections of two patients[31 ] but a clinico-physiologic basis of its existence in the HSCs generating a long-duration,
nearly nonfatigable apogeotropic horizontal PN during SRT was elaborated much later.[6 ]
[7 ]
[29 ] In the last decade, seven prospective interventional studies[32 ]
[33 ]
[34 ]
[35 ]
[36 ]
[37 ]
[38 ] have addressed patients with the apogeotropic variant of HSC-BPPV but only two[34 ]
[36 ] out of these seven include subjects with exclusive HSC-BPPV-cu , while the others[32 ]
[33 ]
[35 ]
[37 ]
[38 ] include those with short anterior arm canalolithiasis as well. Segregation of the
cupulolithiasis (Cup-C or Cup-U), and its influence on the treatment of 40 patients
with ostensive HSC-BPPV-cu with either one-stage (Cup-U) or two-stage (Cup-C) forced prolonged positioning (FPP)
is described by the Chiou et al[32 ] but the study possibly includes patients with short anterior arm horizontal semicircular
canalolithiasis (duration of the apogeotropic horizontal PN and its unchanging character
not specified).
We are reporting a case series of four patients, with HSC-BPPV-cu , who visited our center between June 2019 and July 2020. All four patients were treated
with some form of physical therapy, and the results were audited in the short term
immediately and after an hour. The resolution of vertigo as well as the disappearance
of the apogeotropic PN, during the verifying SRT, was the endpoint of treatment with
physical therapy. To the best of the authors’ knowledge, no such study of patients
diagnosed with HSC-BPPV-cu has been reported from India hitherto.
Material and Methods
The study was approved by the ethics committee of the attending otoneurology center.
The HSC-BPPV-cu was diagnosed as per the following criteria:
Rotational vertigo triggered by changes in the position of the head relative to gravity.
Apogeotropic horizontal PN elicited by the SRT, the side with the weaker nystagmus
was considered pathological as per Ewald’s second law.[39 ]
The duration apogeotropic horizontal PN elicited during the SRT lasted more than a
minute and its attributes (direction and threshold duration) did not change with at
least three times executed SRT.
Vertigo associated with the concomitant elicited PN.
Exclusion criteria were: BPPV treated with any form of physical therapy in the past,
posttraumatic BPPV, a diagnosis of other peripheral vestibular disorders (Meniere’s
disease, vestibular neuritis, vestibular paroxysmia, etc.), and vertigo secondary
to central nervous system disorders.
Informed consent was taken from all four participants. The general physical examination
and vitals of all patients were normal. In all patients, the screening examination
of the cervical spine did not reveal any limitation of movement and the examination
of the back region did not reveal kyphoscoliosis. The lumbosacral spine assessment,
including straight-leg raising (SLR) and reverse SLR tests, were normal in all patients.
The neurological examination revealed normal cranial nerve examination; strength was
grade 5/5 in all four limbs with normal deep tendon reflexes, and bilateral plantar
reflexes were flexor. Examination of the cerebellar system revealed no spontaneous
or gaze-evoked nystagmus, and there was no appendicular or axial incoordination. The
otoneurological examination revealed normal vertical and horizontal saccadic and smooth
pursuit eye movements. The head impulse test was bilaterally normal.
The pathological side is identified by the SRT. The SRT (
[Videos 1 ], [4 ], and
[7 ]
) is done with the patient in long-sitting on the examination table. The patient is
moved to supine with her head landing on a four-inch-thick pillow, so it is anteflexed
to 30 degrees in this position. The supine neutral position is maintained for 30 seconds
to look for lying-down nystagmus (LDN). Thereupon, the patient’s head is rotated first
to one side and maintained until the elicited PN lasts. After the lateral head roll
to one side, the patient’s head is brought to the neutral supine position and then
briskly rolled in the yaw-axis to the other side, and maintained until the elicited
PN lasts. The SRT was performed multiple times (at least three times at an interval
of 5 minutes) in all patients to testify the perseverance of its polarity (apogeotropic)
as well as its long duration (more than a minute). Immediately after a therapeutic
maneuver, and after 1 hour, a verifying SRT was repeated to assess its outcome. The
patients were instructed to report next day in case the vertigo recurs. The recovery
was audited in terms of the disappearance of vertigo, as well as the previously observed
diagnostic apogeotropic horizontal PN. No more than five different therapeutic maneuvers
were performed on any one patient in a single day. The therapeutic positional maneuvers
and the physical therapies used were as under:
HSM (
[Fig. 1 ]
) (
[Videos 2 ], [5 ], and
[8 ]
) is performed with the patient in short-sitting and lower limbs hanging along the
long edge of the examination table. The head is anteflexed 30 degrees in the pitch
plane and briskly shaken by the excursions of 30 degrees side-to-side in the yaw axis
for around 30 seconds. Two sequent HSM are done in one session of treatment. The rapid
acceleration and deceleration during HSM generate inertial forces in the otoconial
debris (irrespective of the side to which it is attached) adherent to the cupula that
causes its detachment or loosens it.
Fig. 1 Head-shaking maneuver (HSM). The head is anteflexed 30 degrees in the pitch plane
and briskly shaken by the excursions of 30 degrees side-to-side at an approximate
rate of 3 Hz in the yaw axis for approximately 30 seconds.
Posterosuperior meatal oscillation with a handheld vibrator (
[Video 10 ]
) is performed with the patient number 4 in the right lateral recumbent position.
Oscillations are delivered to the suprameatal triangle in the posterosuperior area
of the involved left ear with an electrically operated handheld vibrator (Hitachi
Magic Wand with speeds: low 5,000 revolutions per minute [RPM], high 6,000 RPM) for
approximately 60 seconds.
Video 1 During the initial supine roll test, yawing the head of the patient number 1 to the
left, elicited after a latency of 5 seconds stronger apogeotropic horizontal positional
nystagmus that lasted 173 seconds (till the time head remained yawed to the left),
and yawing the head to the right elicited after a latency of 4 seconds, a weaker apogeotropic
horizontal positional nystagmus that lasted 47 seconds.
Video 2 Head-shaking maneuver is performed with the patient number 1 in short-sitting and
lower limbs hanging along the long edge of the examination table. The head is anteflexed
30 degrees in the pitch plane and briskly shaken by the excursions of 30 degrees side-to-side
in the yaw axis for around 30 seconds.
Video 3 The verifying supine roll test of patient number 1 performed 24 hours after the head-shaking
maneuver did not elicit horizontal positional nystagmus on maximal yawing of the head
to the right and left and the patient did not complain of vertigo either.
Video 4 Supine roll test of patient number 2 elicits an apogeotropic horizontal positional
nystagmus on yawing the head maximally to the right as well as to the left, which
is visibly stronger on the right. The latency of the apogeotropic horizontal positional
nystagmus is 4 seconds on either side, and its duration is 190 seconds on the right
and 60 seconds on the left side. The characteristics of the apogeotropic horizontal
positional nystagmus did not change during several cycles of the diagnostic supine
roll test, implying in all probability a pathology of left horizontal semicircular
cupulolithiasis.
Video 5 Head-shaking maneuver is performed with the patient number 2 in short-sitting and
lower limbs hanging along the long edge of the examination table. The head is anteflexed
30 degrees in the pitch plane and briskly shaken by the excursions of 30 degrees side-to-side
in the yaw axis for around 30 seconds.
Video 6 The verifying supine roll test of patient number 2 performed an hour after the head-shaking
maneuver did not elicit any lying down nystagmus or horizontal positional nystagmus
on maximal yawing of the head to the right and left and the patient did not complain
of vertigo either.
Video 7 During the supine roll test, the patient number 4 is moved from long sitting on the
examination table to supine with her head landing on a four-inch-thick pillow resulting
in its 30 degrees anteflexion. In the neutral supine position, lying-down nystagmus
lasting 26 seconds beating to the patient’s left is elicited. Yawing the head to the
left as well as right elicits an apogeotropic horizontal positional nystagmus, which
is visibly stronger on the right. The duration of the apogeotropic horizontal positional
nystagmus is 156 seconds on the right and 60 seconds on the left side. The characteristics
of the apogeotropic horizontal positional nystagmus did not change during several
cycles of the diagnostic supine roll test, implying in all probability a pathology
of left horizontal semicircular cupulolithiasis.
Video 8 Head-shaking maneuver is performed with the patient number 4 in short-sitting and
lower limbs hanging along the long edge of the examination table. The head is anteflexed
about 30 degrees in the pitch plane and briskly shaken by the excursions of 30 degrees
side-to-side in the yaw axis for approximately 30 seconds.
Video 9 Immediately after the head-shaking maneuver is carried out in patient number 4, the
verifying supine roll test elicits a left beating lying-down nystagmus lasting 60
seconds in the neutral supine position. The lateral head rolls to the right as well
as to the left elicit apogeotropic horizontal nystagmus till the time maximal head
yawing is maintained. Elicited apogeotropic horizontal nystagmus is stronger on the
lateral head roll to the right.
Video 10 The right lateral recumbent positioning of the patient number 4 is done. Oscillations
are delivered to the suprameatal triangle in the posterosuperior area of the involved
left ear with an electrically operated handheld vibrator (Hitachi magic wand with
speeds: low 5,000 RPM, high 6,000 RPM) for approximately 60 seconds.
Video 11 Immediately after the left posterosuperior suprameatal oscillation delivered by the
handheld electrical vibrator to patient number 4, the verifying supine roll test elicits
no lying-down nystagmus in the neutral supine position. The lateral head rolls to
the right as well as to the left elicit apogeotropic horizontal nystagmus till the
time maximal head is kept yawed. Elicited apogeotropic horizontal nystagmus is stronger
on the lateral head roll to the right.
Video 12 The patient number 4 is positioned supine with her head in 30 degrees of anteflexion
on a four-inch-thick pillow. The head-rolling maneuver in the supine position is executed.
The head is quickly rolled towards the healthy side, and slowly towards the affected
ear for eight times. By the virtue of the generated inertial forces, quick movement
is expected to detach the otoconial debris adherent to the cupula on its canal side,
and the slow movement is presumed to facilitate the migration of the detached otoconial
debris toward the utricle under the effect of gravitational forces.
180°head-rolling maneuver
[40 ] (
[Fig. 2 ]
,
[Video 12 ]
) is performed with the patient positioned supine with her head in 30 degrees flexion
on a four-inch-thick pillow. The head-rolling maneuver in the supine position is executed.
The head is quickly rolled towards the healthy side and slowly towards the affected
ear eight times. By the generated inertial forces, quick movement is expected to detach
the otoconial debris adherent to the cupula on its canal side, and the slow movement
is presumed to facilitate the migration of the detached otoconial debris toward the
utricle under the effect of gravitational force.
Fig. 2 180-degree head-rolling maneuver in supine recumbent position. With patient in the
supine recumbent position, her head is quickly rolled 180 degrees in the yaw axis
from the diseased left to the healthy right side. Thereupon, it is slowly rolled back
from the healthy right to the diseased left side. Plausibly the quick movement either
detaches away or loosens the otoconial debris adherent to the canal side of the cupula
(Cup-C) under the influence of generated inertial forces. The slow movement facilitates
the migration within the horizontal semicircular canal from its short anterior ampullary
arm to the long posterior nonampullary arm under the influence of gravitational force.
Video 13 Immediately after alternating quick head rolling for a total of eight times from the
diseased left to the healthy right side and slow head rolling from the healthy right
to the diseased left side, the verifying supine roll test of patient number 4 elicits
lying-down nystagmus of 12 seconds duration beating to the patient’s left in the neutral
supine position. The lateral head rolls to the right as well as to the left elicit
apogeotropic horizontal nystagmus of more than a minute duration (till the time maximal
head yawing is maintained). Elicited apogeotropic horizontal nystagmus is stronger
on the lateral head roll to the right.
Video 14 Immediately after forced prolonged positioning (FPP) in the left lateral recumbent
position for 1 hour, the verifying supine roll test in patient number 4 elicits lying-down
nystagmus of 28 seconds duration beating to the patient’s right in the neutral supine
position. The lateral head rolls to the right as well as to the left elicit geotropic
horizontal nystagmus of 25 seconds duration to either side. Elicited geotropic horizontal
nystagmus is stronger on the lateral head roll to the left.
Video 15 Gufoni maneuver for the transformed left geo -horizontal semicircular canal-benign paroxysmal positional vertigo (HSC-BPPV) is
performed by instructing the patient number 4 to be in short siting with both lower
limbs hanging down and briskly moving the patient to the right (contralesional) lateral
recumbent position and maintaining the latter position for 1 minute (step 1). Thereupon,
the patient’s head is rotated approximately 45 degrees downwards in the yaw-axis and
is maintained for 2 minutes (step 2), after which she is positioned upright to the
short sitting.
FPP is carried by instructing the patient number 4 to position the left lateral recumbent
for an hour. The rationale for left lateral recumbent FPP is to detach the otoconial
debris from the canal side of the utricle under the effect of gravitational force
(
[Fig. 3 ]
). The otoconial debris adherent to the canal side of the cupula is plausibly loosened
by the previously executed HSM and 180-degree head-rolling maneuver.
Fig. 3 Forced prolonged positioning (FPP) in left lateral recumbent position for 1 hour.
The patient is positioned left lateral recumbent for 1 hour. In the left lateral recumbent
FPP, the otoconial debris (in red), which has either detached or loosened from the
canal side of the cupula, migrates (blue arrow) within the short ampullary anterior
arm of the canal toward its posterior end.
Gufoni maneuver (
[Video 15 ]
) for the geotropic left HSC-BPPV is performed with the patient number 4 in short
sitting with both lower limbs hanging down the examination table. From short sitting,
she is positioned right (contralesional) lateral recumbent for a minute. Thereupon,
her head is rotated approximately 45 degrees downwards in the yaw-axis and maintained
for 2 minutes, after which she is positioned to the upright sitting.
Results
All four patients included in the study had a history of positionally triggered vertigo
on lying supine, rolling to either of the side lateral positions, and on getting upright
from the supine. The duration of symptoms ranged from 1 to 8 days. The initial diagnostic
SRT elicited apogeotropic horizontal PN of more than a minute duration during the
lateral head rolls in all four patients ([Table 2 ]). The demographic profile, symptom duration, strength of the PN during diagnostic
SRT, LDN direction, diagnosis, therapeutic physical therapy, and results of the verifying
SRT are summarized in [Table 3 ].
Table 2
Characteristics of the apogeotropic horizontal positional nystagmus during the initial
diagnostic supine roll test
Patient number
Diagnostic supine lateral head roll test
Click for video
To right
To left
Latency
Duration
Relative strength
Latency
Duration
Relative strength
Note: The latency, duration, and relative strengths of the apogeotropic horizontal
positional nystagmus on lateral head roll to the right and left during the diagnostic
supine roll test carried initially.
1
4 s
47 s
Weaker
5 s
173 s
Stronger
[Video 1 ]
2
4 s
190 s
Stronger
4 s
60 s
Weaker
[Video 4 ]
3
3 s
79 s
Stronger
5 s
74 s
Weaker
–
4
5 s
156 s
Stronger
18 s
60 s
Weaker
[Video 7 ]
Table 3
Demographic data, symptom duration, diagnostic supine roll test results with localization
(diagnosis) and lateralization, LDN, therapeutic physical therapy, and verifying supine
roll tests (immediately after the physical therapy and at 1 hour)
Patient number
Age
Sex
Vertigo duration
Supine roll test (diagnostic)
Diagnosis
LDN
Physical therapy for treatment
Supine roll test (verifying)
Apogeotropic > 1 min
Immediate
At 1-h
Right
Left
Abbreviations: +, weaker; ++, stronger; apo -HSC-BPPV, apogeotropic horizontal semicircular canal benign paroxysmal positional
vertigo; F, female; HSM, head-shaking maneuver; LDN, lying-down nystagmus; PN, positional
nystagmus.
1.
40
F
1 d
+
++
Right apo -HSC-BPPV
Absent
HSM
No PN
No PN
2.
25
F
8 d
++
+
Left apo -HSC-BPPV
Absent
HSM
No PN
No PN
3.
51
F
2 d
++
+
Left apo -HSC-BPPV
Absent
HSM
No PN
No PN
4.
64
F
4 d
++
+
Left apo -HSC-BPPV
To left
See
[Fig. 4 ]
No PN
No PN
Fig. 4 Management algorithm of patient number 4 with Cup-C variant of left horizontal semicircular
cupulolithiasis.
Patient Number 1, 2, and 3
The patient number 1, 2, and 3 underwent therapeutic HSM (
[Fig. 1 ]
) with their heads flexed approximately 30 degrees. A verifying SRT performed in all
three immediately as well as at 1 hour (
[Videos 3 ]
and
[6 ]
) after the HSM did not elicit apogeotropic horizontal nystagmus, and none of them
had residual vertigo either. A telephonic interview was weekly taken for the next
4 weeks, and all three remained symptom free.
Patient Number 4
The long-duration horizontal apogeotropic PN (
[Video 7 ]
) with perseverance on repeating the supine lateral head roll test suggests HSC-BPPV-cu . Cupulolithiasis of the left HSC is indicated by the side eliciting the weaker horizontal
PN during the supine lateral head roll test as per the Ewald’s second law.[39 ] Accordingly, the patient underwent two sequent HSM (
[Fig. 1 ]
,
[Video 8 ]
). A verifying SRT (
[Video 9 ]
) immediately after the HSM remains unchanged. The right lateral recumbent positioning
of the patient is done. Mastoid oscillations (
[Video 10 ]
) are delivered to the suprameatal triangle in the posterosuperior area of the involved
left ear with an electrically operated handheld vibrator for approximately 60 seconds.
Immediately after the mastoid oscillation, a second verifying SRT (
[Video 11 ]
) remains almost unaltered. A 180-degree head-rolling maneuver (
[Fig. 2 ]
,
[Video 12 ]
) was undertaken. Plausibly the quick movement during the 180-degree head-rolling
maneuver either detaches away or loosens the otoconial debris adherent to the canal
side of the cupula (Cup-C) under the influence of generated inertial forces. The slow
movement facilitates the migration within the HSC from its short anterior arm to the
long posterior arm under the influence of gravitational force. However, a third verifying
SRT (
[Video 13 ]
) immediately after the 180-degree head rolling maneuver remains still unaltered.
Thereupon, the patient is positioned left lateral recumbent for 1 hour (FPP) (
[Fig. 3 ]
). A fourth verifying SRT (
[Video 14 ]
) elicits LDN of 28 seconds duration beating to the patient’s right in the neutral
supine position. The lateral head rolls to the right as well as to the left elicit
geotropic horizontal nystagmus of 25 seconds duration to either side. Elicited geotropic
horizontal nystagmus is stronger on the lateral head roll to the left, indicating
transformation to left long posterior arm horizontal semicircular canalolithiasis.
Two sequent Gufoni maneuvers (
[Video 15 ]
) are undertaken. A fifth verifying SRT (
[Video 16 ]
) performed immediately, and after 1 hour neither elicited the PN nor the patient
had vertigo. A telephonic interview was weekly taken for the next 4 weeks, and the
patient remained symptom free. The treatment protocol of patient number 4 is summarized
in
[Fig. 4 ]
.
Video 16 The verifying supine roll test of the patient number 4 an hour after the Gufoni maneuver
did not elicit any lying-down nystagmus or horizontal positional nystagmus on maximal
yawing of the head to the right and left, and the patient did not complain of vertigo
either.
Discussion
The apogeotropic variant of the HSC-BPPV is attributable to pathologies at three different
sites within the HSC, namely (1) ampullary short anterior arm canalolithiasis, (2)
Cup-U variant of cupulolithiasis, and (3) Cup-C variant of cupulolithiasis. A singular
assessment of the apogeotropic horizontal PN by the SRT is often inadequate to decipher
the precise location of the otoconial debris. The apogeotropic horizontal PN that
either disappears or transforms into the geotropic variant, during SRT, is in all
probability secondary to an unfixed and moveable otoconial debris within the short
anterior arm of the HSC.[24 ]
[41 ]
[42 ]
[43 ]
[44 ] By contrast, apogeotropic horizontal PN secondary to cupulolithiasis (heavy cupula)
invariably lasts more than a minute and remains unaltered even after multiple sequences
of the SRT.[29 ]
[30 ] For this reason, performing multiple sequences of the diagnostic SRT is imperative
to establish a diagnosis of HSC-BPPV-cu , and this was aptly executed in all four cases reported here.
Cases 1 to 3 responded immediately to HSM. Verifying SRT immediately and after 1 hour
did not elicit apogeotropic horizontal PN, and all three remained symptom free over
4 weeks of follow-up on interviewing telephonically. HSM employs the inertia of the
otoconial debris to disengage it from the gelatinous cupula. The offloading of the
otoconial debris from the utricle side of the cupula would bring immediate relief
in vertigo,[23 ] and the detached debris disperses into the gelatinous matrix of the utricle (
[Fig. 1 ]
). The dramatic response in patients 1 to 3 to HSM in terms of alleviation of positionally
triggered vertigo as well as extirpation of the apogeotropic PN (immediately as well
as after 1 hour) explicitly endorse the fact that they suffered from the Cup-U variant
of HSC-BPPV-Cu .
Case 4 failed to respond to the two sequent HSM. Accordingly, we had to recourse to
a series of sequent maneuvers generating the inertial forces to disengage the otoconial
debris adherent to the cupula; mastoid oscillation and 180-degree head-rolling maneuver,
previously reported in the literature.[34 ]
[38 ] Because of the failure of HSM and mastoid oscillation to abolish the apogeotropic
horizontal PN and the concomitant positionally triggered vertigo, it was hypothetically
inferred that otoconial debris is adherent to the canal side of the cupula. The 180-degree
head-rolling maneuver (
[Fig. 2 ]
) and its variants have been used previously with variable success to offload the
otoconial debris purportedly adherent to the canal side of the cupula.[40 ]
[43 ]
[45 ] The 180-degree head-rolling maneuver, and the antecedent HSM and mastoid oscillation
plausibly loosened the otoconial debris adherent to the canal side of the cupula.
A subsequent left lateral recumbent FPP (
[Fig. 3 ]
) for 1 hour just dropped off the otoconial debris loosely adherent to the canal side
of the cupula under the effect of the gravitational force. A verifying SRT executed
just after the FPP elicits an LDN to the patient’s right, and geotropic horizontal
PN on the lateral head roll to either side that was visibly stronger on the left side.
Such a change in the polarity of the horizontal PN from apogeotropic to geotropic
and reversal in the lateralization of its strength (stronger on the lateral head roll
to right initially to stronger on the lateral head roll to left now) indicates transformation
from Cup-C cupulolithiasis to long posterior arm canalolithiasis of the left HSC.
The otoconial debris not only dropped off from the canal side of the cupula after
the left lateral recumbent FPP but also relocated to the posterior arm of the HSC.
Based on our case series, we suggest an algorithm for action when an apogeotropic
horizontal PN is elicited on SRT (
[Fig. 5 ]
).
Fig. 5 Suggested algorithm for action when supine roll test (SRT) elicits apogeotropic horizontal
positional nystagmus.
Conclusion
HSC-BPPV-cu is a distinct disorder of the membranous vestibular labyrinth. Not merely the lateralization
but the identification of the side of the cupula to which otoconial debris is adherent
has a bearing on its management. The physical therapy and maneuvers either employ
the inertial forces generated by the maneuvers (e.g., HSM, mastoid oscillation, and
180-degree head-rolling maneuver) or the gravitational force (e.g., FPP) or a combination
of the two for disengaging the otoconial debris adhering to the gelatinous cupula.
Immediate alleviation of vertigo and disappearance of the apogeotropic horizontal
PN after HSM suggests Cup-U variant of the disorder (cases 1, 2, and 3 ) as the otoconial debris detached from the utricular side of the cupula disperses
in the gelatinous matrix of the utricle. Failure to resolve the positionally triggered
vertigo and nystagmus after HSM may occur when the otoconial debris is either still
tightly adherent to the cupula (on any side) or else in the Cup-C variant in which
disengagement transforms cupulolithiasis into canalolithiasis. A transformation from
Cup-C cupulolithiasis to posterior long arm canalolithiasis (case 4) is attributed
to combining different physical therapies (HSM, mastoid oscillation, 180-degree head-rolling
maneuver, and FPP), and auditing its effect by an immediate SRT.
The American Academy of Otolaryngology, Head, and Neck Surgery Foundation (AAO-HNSF)
clinical published guidelines for BPPV[46 ] found insufficient evidence to recommend a preferred physical therapy for the HSC-BPPV-cu . Future randomized controlled trials are expected to address the segregation of the
cases of apogeotropic HSC-BPPV due to short anterior arm canalolithiasis from those
secondary to cupulolithiasis of the HSC.
