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A novel maneuver for diagnosis and treatment of torsional-vertical down beating positioning nystagmus: anterior canal and apogeotropic posterior canal BPPV

Abstract

Introduction

In patients with benign paroxysmal positional vertigo, BPPV; a torsional-vertical down beating positioning nystagmus can be elicited in the supine straight head-hanging position test or in the Dix-Hallpike test to either side. This type of nystagmus can be explained by either an anterior canal BPPV or by an apogeotropic variant of the contralateral posterior canal BPPV Until now all the therapeutic maneuvers that have been proposed address only one possibility, and without first performing a clear differential diagnosis between them.

Objective

To propose a new maneuver for torsional-vertical down beating positioning nystagmus with a clear lateralization that takes into account both possible diagnoses (anterior canal-BPPV and posterior canal-BPPV).

Methods

A prospective cohort study was conducted on 157 consecutive patients with BPPV. The new maneuver was performed only in those with torsional-vertical down beating positioning nystagmus with clear lateralization.

Results

Twenty patients (12.7%) were diagnosed with a torsional-vertical down beating positioning nystagmus. The maneuver was performed in 10 (6.35%) patients, in whom the affected side was clearly determined. Seven (4.45%) patients were diagnosed with an anterior canal-BPPV and successfully treated. Two (1.25%) patients were diagnosed with a posterior canal-BPPV and successfully treated with an Epley maneuver after its conversion into a geotropic posterior BPPV.

Conclusion

This new maneuver was found to be effective in resolving all the cases of torsional-vertical down beating positioning nystagmus-BPPV caused by an anterior canal-BPPV, and in shifting in a controlled way the posterior canal-BPPV cases of the contralateral side into a geotropic-posterior-BPPV successfully treated during the followup visit. Moreover, this new maneuver helped in the differential diagnosis between anterior canal-BPPV and a contralateral posterior canal-BPPV.

Keywords
Benign paroxysmal positional vertigo; Anterior semicircular canal; Down beating nystagmus; Apogeotropic posterior canal BPPV; Maneuver

Resumo

Introdução

Em pacientes com vertigem posicional paroxística benigna, VPPB, um nistagmo vertical para baixo com componente de torção pode ser provocado no teste head hanging supino executado na posição reta ou no teste de Dix-Hallpike para qualquer um dos lados. Esse tipo de nistagmo pode ser explicado por uma VPPB do canal anterior ou por uma variante apogeotrópica da VPPB do canal posterior contralateral. Até agora, todas as manobras terapêuticas propostas abordam apenas uma possibilidade, sem antes fazer um diagnóstico diferencial claro entre elas.

Objetivo

Propor uma nova manobra para nistagmo vertical para baixo com componente de torção com uma lateralização clara que leve em consideração os dois diagnósticos possíveis, VPPB do canal anterior e VPPB do canal posterior.

Método

Um estudo de coorte prospectivo foi conduzido em 157 pacientes consecutivos com VPPB. A nova manobra foi feita apenas nos pacientes com nistagmo vertical para baixo com componente de torção, com lateralização nítida.

Resultados

Vinte pacientes (12,7%) foram diagnosticados com nistagmo vertical para baixo com componente de torção. A manobra foi feita em 10 (6,35%) pacientes, nos quais o lado afetado foi claramente determinado. Sete (4,45%) pacientes foram diagnosticados com VPPB do canal anterior e tratados com sucesso. Dois (1,25%) pacientes foram diagnosticados com VPPB do canal posterior e tratados com sucesso com a manobra de Epley após sua conversão para VPPB geotrópica de canal posterior.

Conclusão

Essa nova manobra mostrou-se eficaz na resolução de todos os casos de VPPB com nistagmo vertical para baixo com componente de torção causada por VPPB do canal anterior. E na mudança de forma controlada dos casos de VPPB do canal posterior do lado contralateral para uma VPPB geotrópica de canal posterior tratada com sucesso durante a consulta de seguimento. Além disso, essa nova manobra auxiliou no diagnóstico diferencial entre a VPPB do canal anterior e a VPPB do canal posterior contralateral.

Palavras-chave
Vertigem posicional paroxística benigna; Canal semicircular anterior; Nistagmo para baixo; Forma apogeotrópica da VPPB do canal posterior; Manobra

Introduction

Benign paroxysmal positional vertigo (BPPV) is one of the most common and treatable causes of peripheral vertigo, in which one or more semicircular canals are abnormally stimulated by loose otoconia displaced from the otolith organs.11 Argaet EC, Bradshaw AP, Welgampola MS. Benign positional vertigo, its diagnosis, treatment and mimics. Clin Neurophysiol Pract. 2019;4:97-111. Through head movements with respect to gravity, free otoconia activate the semicircular canal afferents, producing a false perception of head rotation and eliciting the corresponding nystagmus.22 Furman JM, Cass SP. Benign paroxysmal positional vertigo. N Engl J Med. 1999;341:1590-1596.

During clinical examination of some patients with a history of BPPV, a nystagmus beating predominantly downward but with a small torsional component in which the upper pole of the eye beats toward the affected ear, called torsional-vertical down beating positioning nystagmus (TVP-DBNy), can be elicited in the supine straight head-hanging (HH) position test or in the Dix-Hallpike (DH) test to either side. In the context of BPPV, that type of nystagmus can be explained by either an anterior canal BPPV (AC-BPPV) or by an apogeotropic variant of the posterior canal BPPV (APC-BPPV).11 Argaet EC, Bradshaw AP, Welgampola MS. Benign positional vertigo, its diagnosis, treatment and mimics. Clin Neurophysiol Pract. 2019;4:97-111.,33 Califano L, Salafia F, Mazzone S, Melillo MG, Califano M. Anterior canal BPPV and apogeotropic posterior canal BPPV: two rare forms of vertical canalolithiasis. Acta Otorhinolaryngol Ital. 2014;34:189-197.

4 Vannucchi P, Pecci R, Giannoni B, Di Giustino F, Santimone R, Mengucci A. Apogeotropic posterior semicircular canal benign paroxysmal positional vertigo: some clinical and therapeutic considerations. Audiol Res. 2015;5:38-43.
-55 Vannucchi P, Pecci R, Giannoni B. Posterior semicircular canal benign paroxysmal positional vertigo presenting with torsional downbeating nystagmus: an apogeotropic variant. Int J Otolaryngol. 2012;2012:2012:413603.

AC-BPPV accounts approximately 3%-12% of all BPPV variants.66 Anagnostou E, Kouzi I, Spengos K. Diagnosis and treatment of anterior-canal benign paroxysmal positional vertigo: a systematic review. J Clin Neurol. 2015;11:262-267.,77 Yang X, Ling X, Shen B, Hong Y, Li K, Si L, et al. Diagnosis strategy and Yacovino maneuver for anterior canal-benign paroxysmal positional vertigo. J Neurol. 2019;266:1674-1684. Although vertigo and nystagmus can be provoked by a DH or a HH test, the latter seems to be the most suitable as the head reaches a more dependent position and the canal is closest to the mid-sagittal plane though extremely curved.77 Yang X, Ling X, Shen B, Hong Y, Li K, Si L, et al. Diagnosis strategy and Yacovino maneuver for anterior canal-benign paroxysmal positional vertigo. J Neurol. 2019;266:1674-1684.

8 Cortés-Domínguez I, Fernández-Seara MA, Pérez-Fernández N, Burguete J. Systematic method for morphological reconstruction of the semicircular canals using a fully automatic skeletonization process. Appl Sci. 2019;9:4904.
-99 Yacovino DA, Hain TC, Gualtieri F. New therapeutic maneuver for anterior canal benign paroxysmal positional vertigo. J Neurol. 2009;256:1851-1855. In the HH position free otoconia in the anterior canal (AC) drift away from the cupula, producing an excitatory response and consequently the clinical examination will reveal a TVP-DBNy. The torsional component is usually less intense than the vertical, probably due to the proximity of the AC to the sagittal plane (41°) when compared to the posterior canal (56°),99 Yacovino DA, Hain TC, Gualtieri F. New therapeutic maneuver for anterior canal benign paroxysmal positional vertigo. J Neurol. 2009;256:1851-1855.,1010 Anagnostou E, Stamboulis E, Kararizou E. Canal conversion after repositioning procedures: comparison of semont and epley maneuver. J Neurol. 2014;261:866-869. and its presence provides a localizing clue.1111 Eggers S.DZ, Bisdorff A, Von Brevern M, Zee DS, Kim J, Perez-Fernandez N, et al. Classification of vestibular signs and examination techniques: nystagmus and nystagmus-like movements: consensus document of the Committee for the International Classification of Vestibular Disorders of the Bárány Society. J Vestib Res Equilib Orientat. 2019;29:57-87. In AC-BPPV, latency is typically absent and the nystagmus is fatigable,33 Califano L, Salafia F, Mazzone S, Melillo MG, Califano M. Anterior canal BPPV and apogeotropic posterior canal BPPV: two rare forms of vertical canalolithiasis. Acta Otorhinolaryngol Ital. 2014;34:189-197. however a short latency and a long duration nystagmus have also been described.44 Vannucchi P, Pecci R, Giannoni B, Di Giustino F, Santimone R, Mengucci A. Apogeotropic posterior semicircular canal benign paroxysmal positional vertigo: some clinical and therapeutic considerations. Audiol Res. 2015;5:38-43. When returning to the sitting position some authors have described a lack inversion of the down-beating vertical nystagmus,33 Califano L, Salafia F, Mazzone S, Melillo MG, Califano M. Anterior canal BPPV and apogeotropic posterior canal BPPV: two rare forms of vertical canalolithiasis. Acta Otorhinolaryngol Ital. 2014;34:189-197.,44 Vannucchi P, Pecci R, Giannoni B, Di Giustino F, Santimone R, Mengucci A. Apogeotropic posterior semicircular canal benign paroxysmal positional vertigo: some clinical and therapeutic considerations. Audiol Res. 2015;5:38-43. while others described an inversion in the 42% of cases.77 Yang X, Ling X, Shen B, Hong Y, Li K, Si L, et al. Diagnosis strategy and Yacovino maneuver for anterior canal-benign paroxysmal positional vertigo. J Neurol. 2019;266:1674-1684.

APC-BPPV accounts approximately 2.5% of BPPV forms.33 Califano L, Salafia F, Mazzone S, Melillo MG, Califano M. Anterior canal BPPV and apogeotropic posterior canal BPPV: two rare forms of vertical canalolithiasis. Acta Otorhinolaryngol Ital. 2014;34:189-197. In this variant of BPPV, loose otoconia are localized in the distal part of the non-ampullary arm of the posterior canal (PC), near the common crus. When HH or DH tests are performed, otoconia move towards the cupula producing an ampullopetal endolymphatic flow and therefore an inhibitory discharge of the posterior ampullary nerve.44 Vannucchi P, Pecci R, Giannoni B, Di Giustino F, Santimone R, Mengucci A. Apogeotropic posterior semicircular canal benign paroxysmal positional vertigo: some clinical and therapeutic considerations. Audiol Res. 2015;5:38-43. The APC-BPPV is characterized by a paroxysmal positioning nystagmus in the opposite direction to the one evoked in typical (geotropic) posterior canal BPPV: a TVP-DBNy. The linear component is down-beating, and the torsional component is towards the unaffected ear (upper pole of the eye beating to the left for the right canal, and to the right for the left canal), similar to contra-lateral AC-BPPV.55 Vannucchi P, Pecci R, Giannoni B. Posterior semicircular canal benign paroxysmal positional vertigo presenting with torsional downbeating nystagmus: an apogeotropic variant. Int J Otolaryngol. 2012;2012:2012:413603. In APC-BPPV the nystagmus is mainly vertical, has no latency, when raising up to the upright position does not always reverse its direction, and the a torsional component is not always clear.33 Califano L, Salafia F, Mazzone S, Melillo MG, Califano M. Anterior canal BPPV and apogeotropic posterior canal BPPV: two rare forms of vertical canalolithiasis. Acta Otorhinolaryngol Ital. 2014;34:189-197.

4 Vannucchi P, Pecci R, Giannoni B, Di Giustino F, Santimone R, Mengucci A. Apogeotropic posterior semicircular canal benign paroxysmal positional vertigo: some clinical and therapeutic considerations. Audiol Res. 2015;5:38-43.
-55 Vannucchi P, Pecci R, Giannoni B. Posterior semicircular canal benign paroxysmal positional vertigo presenting with torsional downbeating nystagmus: an apogeotropic variant. Int J Otolaryngol. 2012;2012:2012:413603.

As previously mentioned TVP-DBNy can be caused by either an AC-BPPV or an APC-BPPV, but in a patient it is very difficult to differentiate the AC from the PC variant and accordingly the side. Until now all the therapeutic maneuvers that have been proposed address only one possibility, and without first performing a clear differential diagnosis between them.44 Vannucchi P, Pecci R, Giannoni B, Di Giustino F, Santimone R, Mengucci A. Apogeotropic posterior semicircular canal benign paroxysmal positional vertigo: some clinical and therapeutic considerations. Audiol Res. 2015;5:38-43.,99 Yacovino DA, Hain TC, Gualtieri F. New therapeutic maneuver for anterior canal benign paroxysmal positional vertigo. J Neurol. 2009;256:1851-1855.,1212 Rahko T. The test and treatment methods of benign paroxysmal positional vertigo and an addition to the management of vertigo due to the superior vestibular canal (BPPV-SC) Clin Otolaryngol Allied Sci. 2002;27:392-395.

13 Kim YK, Shin JE, Chung JW. The effect of canalith repositioning for anterior semicircular canal canalithiasis. ORL. 2005;67:56-60.

14 Honrubia V, Baloh RW, Harris MR, Jacobson KM. Paroxysmal positional vertigo syndrome. Am J Otol. 1999;20:465-470.
-1515 Califano L, Capparuccia PG, Di Maria D, Melillo MG, Villari D. Treatment of benign paroxysmal positional vertigo of posterior semicircular canal by “Quick Liberatory Rotation Manoeuvre”. Acta Otorhinolaryngol Ital. 2003;23:161-167.

Different maneuvers have been proposed for the treatment of AC-BPPV when the pathologic side is identified such as the Rahko maneuver,1212 Rahko T. The test and treatment methods of benign paroxysmal positional vertigo and an addition to the management of vertigo due to the superior vestibular canal (BPPV-SC) Clin Otolaryngol Allied Sci. 2002;27:392-395. the reverse Epley,1414 Honrubia V, Baloh RW, Harris MR, Jacobson KM. Paroxysmal positional vertigo syndrome. Am J Otol. 1999;20:465-470. or the maneuver described by Kim et al.1313 Kim YK, Shin JE, Chung JW. The effect of canalith repositioning for anterior semicircular canal canalithiasis. ORL. 2005;67:56-60. In 2009, Yacovino and Hain proposed a maneuver that works symmetrically for both anterior canals, regardless of which side is affected.99 Yacovino DA, Hain TC, Gualtieri F. New therapeutic maneuver for anterior canal benign paroxysmal positional vertigo. J Neurol. 2009;256:1851-1855. On the other hand, the literature is scarce regarding the treatment of APC-BPPV. Califano et al. in 2014 described the use of a maneuver named “quick liberatory rotation maneuver”, which is similar to the Epley maneuver with the difference that the rotation of the head in the horizontal plane is followed by the trunk, and also movements are carried out very quickly.1515 Califano L, Capparuccia PG, Di Maria D, Melillo MG, Villari D. Treatment of benign paroxysmal positional vertigo of posterior semicircular canal by “Quick Liberatory Rotation Manoeuvre”. Acta Otorhinolaryngol Ital. 2003;23:161-167. Also, Vannucchi proposed a maneuver in 2015 based on the Semont maneuver named as “Demi-Semont”.44 Vannucchi P, Pecci R, Giannoni B, Di Giustino F, Santimone R, Mengucci A. Apogeotropic posterior semicircular canal benign paroxysmal positional vertigo: some clinical and therapeutic considerations. Audiol Res. 2015;5:38-43.

We propose here a new maneuver for TVP-DBNy when it is possible to determine the affected side. This maneuver is designed to remove the debris through the common crus in AC-BPPV; but in case of contralateral APC-BPPV, the same maneuver will shift in a controlled way the debris from the non-ampullary arm into the ampullary arm of the contralateral posterior canal, turning it into a geotropic posterior anal BPPV (PC-BPPV).

Methods

Patients and methods

A prospective cohort study was conducted on 157 consecutive patients diagnosed with BPPV during three consecutive months (December 2019-February 2020) in the neurotology clinic of our department. Only patients with TVP-DBNy and diagnosed with an AC-BPPV using the diagnostic criteria established by the Bárány Society in 2015 were included in the analysis.1616 Von Brevern M, Bertholon P, Brandt T, Fife T, Imai T, Nuti D, et al. Benign paroxysmal positional vertigo: diagnostic criteria. J Vestib Res Equilib Orientat. 2015;25:105-117.

All patients underwent a routine neurotologic examination that included eye position, ocular motility, spontaneous and gaze-evoked nystagmus with and without visual fixation, headshaking-induced nystagmus or vibration-induced nystagmus and the clinical visual suppression and visual-vestibular interaction tests. The VOR was tested both clinically and with the video head-impulse test (the 6 semicircular canals). Dynamic position tests were orderly performed first the HH position test, then the supine, left and right lateral and finally the DH test. All tests were assessed and recorded using a videonystagmograph (VNG) system (VideoFrenzel Interacoustics VF505m) except the DH test, for which common Frenzel goggles were used. Audiometry was performed when indicated and all subjects were evaluated with a brain MRI.

The exclusion criteria were multicanal BPPV and central vestibular pathology according to findings in the clinical exam or MRI.1717 Esteban-Sánchez J, Rueda-Marcos A, Sanz-Fernández R, Martín-Sanz E. Inferior vertical nystagmus: is magnetic resonance imaging mandatory? Rev Neurol. 2016;62:107-112. This was a high resolution thin-slice temporal bone MRI with special focus on brainstem and cerebellum. Institutional review board approval and informed consent from all patients were obtained.

Particle repositioning maneuver

Our repositioning maneuver is illustrated in Fig. 1. In order to perform this maneuver, and decide the “affected side”, we assumed in all cases that TVP-DBNy was triggered by an AC-BPPV. This maneuver is designed to use both inertial and gravitational force to move debris though the canal. The following steps were systematically followed:

Figure 1
Novel maneuver for the torsional-vertical down beating positioning nystagmus (TVP-DBNy) in patients with BPPV. In this example during the clinical examination, the patient revealed a DBN (Down Beating nystagmus) with a RBT (torsional component the upper pole of the eye beating towards the right or counterclockwise from examiner’s perspective). TVP-DBNy is assumed to be triggered by an AC-BPPV on the right side (Otoconia position and movement are highlight in yellow) or an APC-BPPV on the left side (Otoconia position and movement are highlight in blue). During all the steps of the maneuver, the nystagmus remains the same, because the otoconia and the endolymphatic flow follows the same direction and therefore the same response (ampullofugal/excitatory in AC-BPPV or ampullopetal/inhibitory in APC-BPPV). A and B (Step I), C (Step II), D (Step III), E (Step IV), F and G (Step V), DBN.

Step I: The maneuver starts with the patient in the sitting position with the back vertical at 0° and the head bowed approximately a 45° angle (Fig. 1Aa), Next, the patient’s head is rotated towards the unaffected side (Fig. 1Ba). This position places the AC of the affected side, and the PC of the unaffected in the sagittal plane. (Fig. 1Bb-Bc).

Step II: The patient is quickly bent forward 90° through a flexion of the lumbar spine and hips until the head reaches the height of the knees (Fig. 1Ca). With this movement, the inner ear labyrinth will be at a 90° forward bending;1818 Iglebekk W, Tjell C, Borenstein P. Can the bending forward test be used to detect a diseased anterior semi-circular canal in patients with chronic vestibular multi-canalicular canalithiasis (BPPV)? Acta Otolaryngol. 2019;139:1067-1076. if we add the 45° of head forward flexion Step I, the cupula of the AC will now be at 135° forward bending.1818 Iglebekk W, Tjell C, Borenstein P. Can the bending forward test be used to detect a diseased anterior semi-circular canal in patients with chronic vestibular multi-canalicular canalithiasis (BPPV)? Acta Otolaryngol. 2019;139:1067-1076.

19 Rabbitt RD. Semicircular canal biomechanics in health and disease. J Neurophysiol. 2019;121:732-755.
-2020 Iglebekk W, Tjell C, Borenstein P. Treatment of chronic canalithiasis can be beneficial for patients with vertigo/dizziness and chronic musculoskeletal pain, including whiplash related pain. Scand J Pain. 2015;8:1-7. (Fig. 2) In this position, loose otoconia in the AC will be displaced towards the lowest gravitational point of the canal, first using the inertia generated by the deceleration of the movement and then by gravity (Fig. 1Cb). In the case of a contralateral APC-BPPV, loose otoconia will be displaced towards the lower gravitational point of the non-ampullary arm of the PC by the same mechanisms (Fig. 1Cc). This position is maintained for 1 min.

Figure 2
Right anterior canal spatial positioning during Stage I and II. A, Patient’s head in sitting position (0°). B, Patient’s head after forward flexion (45°). C, Patients head after rotation towards the left side and flexion of the lumbar spine and hips (90°). The right anterior canal cupula reaches approximately a 135° forward rotation.

Step III: The patient’s head is slowly rotated towards the affected side (Fig. 1Da). This rotation induces the displacement of otoconia by gravity towards the common crus (Fig. 1Db). In the case of a contralateral APC-BPPV, the otoconia will reach the lowest gravitational point of the canal (Fig. 1Dc). This position is maintained for 1 min.

Step IV: With the head rotated towards the affected side, the patient is now brought to the sitting position with vertebral column at 0° in a single rapid movement (Fig. 1. Ea). Now the otoconia will be displaced into the common crus (Fig. 1 Eb). In the case of a contralateral APC-BPPV, they will reach the ampullary arm of the posterior canal (Fig. 1Ec).

Step V: The patient’s head is slowly rotated towards the midline (Fig. 1Fa) and is then elevated into a neutral position (Fig. 1Ga). Loose otoconia will be moved out into the utricle (Fig. 1Gb), and in the case of a contralateral APC-BPPV, will get closer to the ampulla of the PC (Fig. 1Gc).

Results

In the study period 20 patients (12.7%) were diagnosed TVP-DBNy. The maneuver was performed in 10 (50%) patients, in whom the affected side was clearly determined (Fig. 3). Nine were females (90%) and one was male (10%), the mean age was 59 years, and 7 (70%) had a previous history of BPPV. Among the patients with previous history of BPPV, 5 (71.4%) remained asymptomatic since the last episode/maneuver (mean of 21.25 months). Two patients (28.6%) remained partially symptomatic since the last episode/maneuver. Patient nº 2 had a right Epley maneuver performed 7 days before and patient nº 9 had also a right Epley maneuver performed 15 days before the assessment.

Figure 3
Flowchart illustrating the study results. TVP-DBNy, Torsional-Vertical Down Beating Positioning Nystagmus; AC-BPPV, Anterior Canal BPPV; APC, Apogeotropic variant of Posterior Canal BPPV.

In the HH position down beating nystagmus was seen in 9 patients with different direction in the torsional component except for 1 (patient nº 4). In 4/9 patients the same nystagmus was seen in the HH test and both right and left DH tests (patients nº 1, nº 7, nº 8, and nº 9); in 3/9 patients the HH test finding was similar when the DH test was performed on just one side (2 were the affected and 1 in the non-affected side) (patients nº 2, nº 5, and nº 10); In 2/9 patients nystagmus was seen only the HH or DH test respectively (patients nº 3 and nº 4). In 1/10 patient (patient nº 4) the HH test was negative and the finding was obtained while performing the left sided DH test. Reversal of nystagmus upon siting was observed in 3 patients (30%) during DH and/or HH tests (Table 1).

Table 1
Clinical characteristics of patients with torsional vertical down beating positioning nystagmus BPPV.

Of the 10 patients here included, 1 was lost to followup. Five (55.56%) patients improved their symptoms and the nystagmus disappeared in the first medical followup (mean time to followup: 5.4 days). In 2 (22.22%) patients, symptoms and the same nystagmus (TVP-DBNy) persisted in the followup visit. A new maneuver was performed: patient nº 7 required a second maneuver and patient nº 8 a third maneuver until their symptoms and the nystagmus disappeared. In two patients (22.22%) at followup symptoms persisted but nystagmus was seen only in the DH test now when performed on the contralateral side, and accordingly were diagnosed of an APC-BPPV (patients nº 7 and nº 8). As in followup nystagmus was now consistent with a canalolithiasis in the ampullar arm, an Epley maneuver was successfully performed in both cases.

Discussion

In our work, we have described a novel method to treat AC-BPPV, which also may be useful to carry out the differential diagnosis between AC-BPPV and the APC-BPPV variant. Both possible diagnoses were taken into account when designing the maneuver.

This study has some limitations. The main limitation is that the variants here considered are unusual and, in the case of the AC-BPPV, even controversial. Down-beat nystagmus is not an infrequent finding when complete vestibular examination is performed in patients with vertigo. Previous authors have noted that this type of nystagmus is linked to BPPV in approximately 50% of the patients,1717 Esteban-Sánchez J, Rueda-Marcos A, Sanz-Fernández R, Martín-Sanz E. Inferior vertical nystagmus: is magnetic resonance imaging mandatory? Rev Neurol. 2016;62:107-112. and that it needs a high index of suspicion to achieve a final diagnosis. In our study down-beat nystagmus was the only finding in 12.7% of patients seen for a confirmed BPPV. This is probably related to the sequence of the examination, which we always began with the HH test. A second limitation was that in order to be able to perform this maneuver, the affected side must be identified; that has been shown to occur in 33%-78% of patients.2121 Casani AP, Cerchiai N, Dallan I, Sellari-Franceschini S. Anterior canal lithiasis: diagnosis and treatment. Otolaryngol Head Neck Surg. 2011;144:412-418.,2222 Lopez-Escamez JA, Molina MI, Gamiz MJ. Anterior semicircular canal benign paroxysmal positional vertigo and positional downbeating nystagmus. Am J Otolaryngol - Head Neck Med Surg. 2006;27:173-178. In the present study, among 20 patients with TVP-DBNy the affected side was identified in only 10 (50%) of them.

With regard to our study population, the results of the clinical examination similar to the ones described by Yang et al.77 Yang X, Ling X, Shen B, Hong Y, Li K, Si L, et al. Diagnosis strategy and Yacovino maneuver for anterior canal-benign paroxysmal positional vertigo. J Neurol. 2019;266:1674-1684. in 40 patients with AC-BPPV. Yang described that nystagmus was obtained in all patients in the HH test (down-beat nystagmus with/without torsional component) and in 42.5% during bilateral DH tests, and that nystagmus reversed direction upon sitting up in 42.5% of patients. In our study those values were 90%, 40% and 30% respectively.

Regarding the frequency of the nystagmus and differential diagnosis some comments are needed. During a clinical evaluation, elucidating whether the presence of TVP-DBNy in patients with a typical history of BPPV is caused either by an AC-BPPV or an APC-BPPV on the contralateral side is very difficult and unlikely to happen in practice. Califano proposed a differential diagnosis between both etiologies based on the clinical examination.33 Califano L, Salafia F, Mazzone S, Melillo MG, Califano M. Anterior canal BPPV and apogeotropic posterior canal BPPV: two rare forms of vertical canalolithiasis. Acta Otorhinolaryngol Ital. 2014;34:189-197. Besides the torsional component to differentiate a right from left AC-BPPV, and a right from left APC-BPPV, he defined the diagnosis of AC-BPPV as certain when the nystagmus was evoked through the HH test and sometimes through the DH test, and the diagnosis of APC-BPPV as certain when the nystagmus was evoked through the DH test and sometimes through the HH test. This approach might be somewhat inaccurate because previous studies have described that both etiologies can be triggered through both tests,55 Vannucchi P, Pecci R, Giannoni B. Posterior semicircular canal benign paroxysmal positional vertigo presenting with torsional downbeating nystagmus: an apogeotropic variant. Int J Otolaryngol. 2012;2012:2012:413603.,77 Yang X, Ling X, Shen B, Hong Y, Li K, Si L, et al. Diagnosis strategy and Yacovino maneuver for anterior canal-benign paroxysmal positional vertigo. J Neurol. 2019;266:1674-1684.,99 Yacovino DA, Hain TC, Gualtieri F. New therapeutic maneuver for anterior canal benign paroxysmal positional vertigo. J Neurol. 2009;256:1851-1855.,2323 Bertholon P, Bronstein AM, Davies RA, Rudge P, Thilo KV. Positional down beating nystagmus in 50 patients: Cerebellar disorders and possible anterior semicircular canalithiasis. J Neurol Neurosurg Psychiatry. 2002;72:366-372. and this is consistent with our results. But what is most important, and the key element in order to differentiate both etiologies, is how to distinguish a right AC-BBPV from a left APC-BPPV and vice versa, and that was not accurately defined by Califano. We propose that the best way to approach a TVP-DBNy is to consider both variants as possible triggers, and this strategy can be achieved with our proposed maneuver.

In our study confirmed AC-BPPV (after the maneuver) was found to be 4.45%, similar to the incidence describe by other authors which is approximately between 3% and 5%.66 Anagnostou E, Kouzi I, Spengos K. Diagnosis and treatment of anterior-canal benign paroxysmal positional vertigo: a systematic review. J Clin Neurol. 2015;11:262-267.,2424 Marques PS, Castillo R, Santos M, Perez-Fernandez N. Repositioning nystagmus: prognostic usefulness? Acta Otolaryngol. 2014;134:491-496. However, if we include the cases in which the etiology of the TVP-DBNy could not be determined (1 of them lost followup and 10 with unclear lateralization), the incidence increases to 11.45%. If we assume those cases as AC-BPPV, the incidence could also be similar to higher incidences described, such as 12.1%.77 Yang X, Ling X, Shen B, Hong Y, Li K, Si L, et al. Diagnosis strategy and Yacovino maneuver for anterior canal-benign paroxysmal positional vertigo. J Neurol. 2019;266:1674-1684. On the other, the incidence of confirmed APC-BPPV (after the maneuver) was found to be 1.27%. It is important to highlight that the diagnosis was made after the maneuver, which converted in a controlled way the APC-BPPV cases into PC-BPPV. Vannucchi described an incidence of 6.25% of APC-BPPV, and also emphasized the misdiagnosis of this variant as an AC-BPPV, when he noted that patients diagnosed witch AC-BPPV would come back to the next checkup with a typical PC-BPPV of the opposite side.55 Vannucchi P, Pecci R, Giannoni B. Posterior semicircular canal benign paroxysmal positional vertigo presenting with torsional downbeating nystagmus: an apogeotropic variant. Int J Otolaryngol. 2012;2012:2012:413603. Our APC-BPPV incidence could be 8.27% if we include the rest of the cases with unclear lateralization. Therefore, one question that results from this study is whether we are presently making an accurate diagnosis of AC-BPPV, or we are just assuming that all TVP-DBNy are caused by an AC-BPPV. It is interesting to highlight that two patients underwent replacement maneuvers prior to proposed maneuver (patients nº 2 and nº 9). The partial persistence of symptoms and the short time since the last maneuver could suggest that these atypical forms of BPPV could be considered as a complication of a previous maneuver.

Several manual reduction methods have been described for treating AC-BPPV when the affected side is identified, such as Rahko,1212 Rahko T. The test and treatment methods of benign paroxysmal positional vertigo and an addition to the management of vertigo due to the superior vestibular canal (BPPV-SC) Clin Otolaryngol Allied Sci. 2002;27:392-395. the reverse Epley1414 Honrubia V, Baloh RW, Harris MR, Jacobson KM. Paroxysmal positional vertigo syndrome. Am J Otol. 1999;20:465-470. or Kim1313 Kim YK, Shin JE, Chung JW. The effect of canalith repositioning for anterior semicircular canal canalithiasis. ORL. 2005;67:56-60. maneuver. All of these have been designed without taking into account a possible APC-BPPV in the contralateral ear, and except for Kim´s maneuver, there are no published data concerning their efficacy. Also, these maneuvers are performed in a supine position, and in some parts of the procedure, the head is in a hanging position, and it is turned in a manner similar to the way PC-BPPV is treated.1212 Rahko T. The test and treatment methods of benign paroxysmal positional vertigo and an addition to the management of vertigo due to the superior vestibular canal (BPPV-SC) Clin Otolaryngol Allied Sci. 2002;27:392-395.

13 Kim YK, Shin JE, Chung JW. The effect of canalith repositioning for anterior semicircular canal canalithiasis. ORL. 2005;67:56-60.
-1414 Honrubia V, Baloh RW, Harris MR, Jacobson KM. Paroxysmal positional vertigo syndrome. Am J Otol. 1999;20:465-470. The AC follows a different trajectory from the PC, therefore maneuvers to treat AC-BPPV must necessarily differ geometrically from those described for the PC.99 Yacovino DA, Hain TC, Gualtieri F. New therapeutic maneuver for anterior canal benign paroxysmal positional vertigo. J Neurol. 2009;256:1851-1855. Since the PC is the most stimulated and assessed in the DH test, it is relevant to assume that the AC is better stimulated and assessed during a bending forward movement.1818 Iglebekk W, Tjell C, Borenstein P. Can the bending forward test be used to detect a diseased anterior semi-circular canal in patients with chronic vestibular multi-canalicular canalithiasis (BPPV)? Acta Otolaryngol. 2019;139:1067-1076. The Yacovino-Hain maneuver works symmetrically for unilateral AC-BPPV, regardless of which side is affected.99 Yacovino DA, Hain TC, Gualtieri F. New therapeutic maneuver for anterior canal benign paroxysmal positional vertigo. J Neurol. 2009;256:1851-1855. If we do not take into account the affected ear, TVP-DBNy can be triggered by loose otoconia in four semicircular canals: both ACs (AC-BPPV) and both PCs (APC-BPPV). By performing this maneuver we are assuming that the problem is in only one AC, and even if both ACs were affected, we are blindly hoping to liberate loose otoconia without knowing exactly where they are, since they could be in any of the four canals. Like the previous ones, the Yacovino-Hain maneuvers do not take into account the possibility of APC-BPPV involvement, and uncontrolled conversions into a PC-BPPV after performing the maneuver have been described.33 Califano L, Salafia F, Mazzone S, Melillo MG, Califano M. Anterior canal BPPV and apogeotropic posterior canal BPPV: two rare forms of vertical canalolithiasis. Acta Otorhinolaryngol Ital. 2014;34:189-197.,77 Yang X, Ling X, Shen B, Hong Y, Li K, Si L, et al. Diagnosis strategy and Yacovino maneuver for anterior canal-benign paroxysmal positional vertigo. J Neurol. 2019;266:1674-1684. Its success rate is between 44.4% and 84.6%,77 Yang X, Ling X, Shen B, Hong Y, Li K, Si L, et al. Diagnosis strategy and Yacovino maneuver for anterior canal-benign paroxysmal positional vertigo. J Neurol. 2019;266:1674-1684.,2525 Chen D, Xiong S, Cui Y. Treatment of anterior canal benign paroxysmal positional vertigo by Yacovino repositioning maneuver. Lin Chung Er Bi Yan Hou Tou Jing Wai Ke Za Zhi. 2015;29:17-19. and some patients will need an additional maneuver to achieve a clinical resolution.33 Califano L, Salafia F, Mazzone S, Melillo MG, Califano M. Anterior canal BPPV and apogeotropic posterior canal BPPV: two rare forms of vertical canalolithiasis. Acta Otorhinolaryngol Ital. 2014;34:189-197.,77 Yang X, Ling X, Shen B, Hong Y, Li K, Si L, et al. Diagnosis strategy and Yacovino maneuver for anterior canal-benign paroxysmal positional vertigo. J Neurol. 2019;266:1674-1684.,99 Yacovino DA, Hain TC, Gualtieri F. New therapeutic maneuver for anterior canal benign paroxysmal positional vertigo. J Neurol. 2009;256:1851-1855. We also recommend to repeat our maneuver until either symptoms and nystagmus disappear, or until a PC-BPPV in the contralateral side is diagnosed. The proposed maneuver might be difficult to carry out in obese patients, nevertheless it can be a good option for elderly patients and those with limitation of neck mobility.

When it comes to the posterior canal BPPV, we have to take into account very unusual variants, such as the “short arm” canalithiasis.2626 Oas JG. Benign paroxysmal positional vertigo. Ann N Y Acad Sci. 2006;942:201-209. In this hypothetical variant, during the DH test, there is either no movement of the otoconia debris or one in an ampullofugal direction. While in the HH test, otoconia movement generates an ampullopetal endolymphatic flow with reversal upon sitting (even this could be the only registered effect) similar to an APC-BPPV.2727 Büki B, Mandalà M, Nuti D. Typical and atypical benign paroxysmal positional vertigo: literature review and new theoretical considerations. J Vestib Res Equilib Orientat. 2014;24(5-6):415-423.,2828 Fath L, Vuong-Chaney H, Rohmer D, Lamy M, Baumgartner D, Simon F, et al. Unusual locations of benign paroxysmal positional vertigo. Otol Neurotol. 2020;41:e735-e743. In this (otherwise hypothetical) situation, the second step of our maneuver (Fig. 4) would transfer the otoconia from the short arm towards the utricle, following the same principle of the Ostrich maneuver described by Fath et al. for the PC “short arm” canalithiasis.2828 Fath L, Vuong-Chaney H, Rohmer D, Lamy M, Baumgartner D, Simon F, et al. Unusual locations of benign paroxysmal positional vertigo. Otol Neurotol. 2020;41:e735-e743.

Figure 4
Otoconia movement in a Posterior Canal “short arm” canalithiasis during the second step of the maneuver.

The bend forward test has been recently described as a diagnostic tool in the assessment of AC-BPPV by Iglebekk et al.1818 Iglebekk W, Tjell C, Borenstein P. Can the bending forward test be used to detect a diseased anterior semi-circular canal in patients with chronic vestibular multi-canalicular canalithiasis (BPPV)? Acta Otolaryngol. 2019;139:1067-1076. The same group described a maneuver called “forward somersault” maneuver,2020 Iglebekk W, Tjell C, Borenstein P. Treatment of chronic canalithiasis can be beneficial for patients with vertigo/dizziness and chronic musculoskeletal pain, including whiplash related pain. Scand J Pain. 2015;8:1-7. in which the patient is turned 360° forwardly. Similarly, to the Yacovino-Hain maneuver it works for both sides, but it does not take into account a possible APC-BPPV Additionally it requires a specialized movable chair and is therefore unlikely to be very clinically practical.

Although this is a small and uncontrolled cohort study, this new maneuver was effective in resolving all the cases of TVP-DBNy caused by an AC-BPPV, and in shifting in a controlled way the APC-BPPV cases of the contralateral side into a geotropic-posterior-BPPV successfully treated during the followup visit. This maneuver could be useful in patients with a TVP-DBNy with a clear lateralization, as an alternative to the Yacovino-Hain maneuver.

  • Peer Review under the responsibility of Associação Brasileira de Otorrinolaringologia e Cirurgia Cérvico-Facial.

Acknowledgements

To Esteban Župan for the help provided in the design of the illustrations.

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Publication Dates

  • Publication in this collection
    17 Oct 2022
  • Date of issue
    Sep-Oct 2022

History

  • Received
    2 July 2020
  • Accepted
    22 Sept 2020
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