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Granddaughter's somersault treats cupulolithiasis of the horizontal semicircular canal
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American Journal of Otolaryngology–Head and Neck Medicine and Surgery 34 (2013) 72 – 74 www.elsevier.com/locate/amjoto
Case report
Granddaughter's somersault treats cupulolithiasis of the horizontal semicircular canal Dirk Czesnik, MD⁎, David Liebetanz, MD Department of Clinical Neurophysiology, Medical School Göttingen, Georg-August-University of Göttingen, Robert-Koch-Str. 40, Göttingen, Germany Revised 22 June 2012
Abstract
We report on a 61-year-old woman with cupulolithiasis of the right horizontal semicircular canal, which is usually difficult to treat. The patient reported that several years ago, similar symptoms relieved completely after having performed several somersaults together with her granddaughter. This time, repetitive somersaults were again effective to treat her benign paroxysmal positional vertigo. Acceleration during a somersault may induce an intracanalicular force strong enough to detach otoconia debris from the cupula. Rolling may then promote their reentrance into the utricle. This case suggests that repetitive somersaults may be an alternative treatment of cupulolithiasis of the horizontal semicircular canal. © 2013 Elsevier Inc. All rights reserved.
1. Introduction Paroxysmal dizziness is commonly caused by benign paroxysmal positional vertigo (BPPV). In most cases of BPPV, the posterior semicircular canal is affected. To a lower extent (5%–10%), the horizontal semicircular canal is involved [1–3]. The BPPV of the horizontal semicircular canal (h-BPPV) typically occurs during turning of the head while lying in a supine position. The etiology of h-BPPV can be variable. In most cases, it is idiopathic, but it can also occur posttraumatically or be induced by positioning maneuvers to treat PBPPV [4]. Clinically, h-BPPV is characterized by a bidirectional horizontal nystagmus, which is triggered as the head of the supine patient is turned from side to side. There are 2 subtypes of h-BPPV: (i) the canalolithiasis is caused by fragments of otoconia that move freely in the horizontal semicircular canal. In case of canalolithiasis, nystagmus is geotropic (toward the undermost ear) and more intense after rotation of the head toward the affected side. It is caused by the movement of otoconial debris within the long arm of the
⁎ Corresponding author. Department of Clinical Neurophysiology, Medical School Göttingen, Robert-koch-Str. 40, Göttingen. Tel.: +49 551 396650; fax: +49 551 3913614. E-mail address: [email protected] (D. Czesnik). 0196-0709/$ – see front matter © 2013 Elsevier Inc. All rights reserved. http://dx.doi.org/10.1016/j.amjoto.2012.07.002
horizontal semicircular canal, which stimulates utriculopetal endolymph flow. The vertigo is triggered by circular acceleration of the horizontal semicircular canal (ii), if cupulolithiasis otoconia are attached to the cupula or are trapped in the proximal segment of the canal near to the cupula. As the head of the supine patient is turned slowly from side to side, the cupula is displaced, which induces asymmetric vestibular stimulation [5,6]. The vertigo typically occurs when the head changes position; circular acceleration does not trigger vertigo. The nystagmus during cupulolithiasis of the horizontal semicircular canal is pathognomonically apogeotropic (away from the undermost ear) and more intense after rotation of the head toward the unaffected ear. Interestingly, there is no effective maneuver available for the treatment of the cupulolithiasis of the horizontal semicircular canal, to date. Here, we suggest that somersaulting may be a more effective treatment maneuver, which relieves symptoms within a few days. 2. Case report The 61-year-old woman turned to the emergency hospitalization because of rotary dizziness, which suddenly occurred after turning in bed that morning. The symptoms were accompanied by nausea and emesis. Several years ago, the patient experienced similar symptoms, which did not
D. Czesnik, D. Liebetanz / American Journal of Otolaryngology–Head and Neck Medicine and Surgery 34 (2013) 72–74
respond to conventional treatment, but remarkably disappeared after having performed several somersaults together with her granddaughter. Preexisting conditions are otosclerosis (surgery in 2000 and 2003) and migraine. The migraine is treated with sumatriptane (100 mg) and mergentane (50 mg) if required. 2.1. Neurologic examination Clinical examination revealed an apogeotropic nystagmus when moving her head left and right while lying in a supine position (see video). The nystagmus was more intense when the head was rotated to the left side. There were no pathological findings in any other cranial nerve. Sensory and motor examination, tendon reflexes, gait, and coordination showed no signs of pathology. Physical examination of the heart, lungs, and abdomen were, accordingly, without pathological findings. The typical history and clinical findings led to the diagnosis of the cupulolithiasis of the h-BPPV with the right horizontal semicircular canal affected. Other reasons for the dizziness were excluded. Because somersault had been previously therapeutically effective, we advised her to somersault repetitively again. Interestingly, only a few somersaults during hospitalization were necessary to decrease the symptoms remarkably and reduce nystagmus. After hospital discharge, we advised repeating the somersaults at home daily. A 6-week
73
follow-up, examination showed no remaining symptoms and no positional nystagmus.
3. Discussion The horizontal semicircular canal (HSC) variant of BPPV is the second most common cause of BPPV. However, so far there is no effective maneuver to treat the cupulolithiasis variant of HSC-BPPV. The most difficult challenge is to detach the otoconia from the cupula. The Lempert or Barbecue maneuver [7] and the forced prolonged position (FPP) [8–11] are the most common maneuvers to treat canalolithiasis. They all aim to reposition of the otoconia out of the semicircular canal. Controlled studies demonstrated an effectiveness of reposition of 75% and 75% to 90% for the Barbecue and FPP maneuvers, respectively. However, the effectiveness of cupulolithiasis treatment by these maneuvers is much lower because they are not likely to detach the otoconia from the cupula [8,12]. Boleas-Aguirre and coworkers [12] reported that only the combination and repetition of FPP or Lempert over 2 weeks were able to increase the effectiveness. However, the suggested FPP for more than 12 hours daily for at least 2 weeks often led to a reduced treatment compliance. In our case, repetitive somersaults reduced the symptoms significantly and effectively within a few days. Acceleration
Fig. 1. Body rotation (A) and rotation of the horizontal semicircular canal during a somersault (B; lateral view). The gray line illustrates the position of horizontal semicircular canal. (C) The left picture illustrates how acceleration in the forward-facing direction during the somersault induce an intracanalicular force strong enough to detach otoconia debris from the cupula. The right picture demonstrates how ongoing rolling may move loose otoconia debris back into the utricle.
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D. Czesnik, D. Liebetanz / American Journal of Otolaryngology–Head and Neck Medicine and Surgery 34 (2013) 72–74
in the forward-facing direction during the somersault may induce an intracanalicular force strong enough to detach otoconia debris from the cupula. The ongoing rolling may move loose otoconia debris back into the utricle (Fig. 1). This case suggests that repetitive somersaulting may be an alternative treatment of the HSC variant of BPPV. To evaluate the effectiveness, a prospective study in more patients is needed. Supplementary data to this article can be found online at http://dx.doi.org/10.1016/j.amjoto.2012.07.002. References [1] Pagnini P, Nuti D, Vannucchi P. Benign paroxysmal vertigo of the horizontal canal. ORL J Otorhinolaryngol Relat spec 1989;51:161-70. [2] McClure JA. Horizontal canal BPV. J Otolaryngol 1985;14:30-5. [3] Cakir BO, Ercan IC, Akir ZAC, Enol C, Sayin I, Turgut S. What is the incidence of horizontal semicircular canal benign paroxysmal positional vertigo? Otolaryngol Head Neck Surg 2006;134:451-4. [4] Baloh RW, Jacobson K, Honrubia V. Horizontal semicircular canal variant of benign positional nystagmus? Neurology 1993;43:2542-9.
[5] Fife TD. Recognition and management of horizontal canal benign positional vertigo. Am J Otol 1998;19:345-51. [6] Nuti D, Vannucchi P, Pagnini P. Benign paroxysmal positional vertigo of the horizontal canal: a form of canalolithiasis with variable clinical features. J Vestib Res 1996;6:173-84. [7] Lempert T, Tiel-Wilck K. A positional maneuver for treatment of horizontal canal benign positional vértigo. Laryngoscope 1996;106: 476-8. [8] Chiou WY, Lee HL, Tsai SC, Yu TH, Lee XX. A single therapy for all subtypes of horizontal canal positional vertigo. Laryngoscope 2005; 115:1432-5. [9] Cassani AP, Vannucci G, Fattori B, Berretini S. The treatment of horizontal canal positional vertigo: our experience in 66 cases. Laryngoscope 2002;112:172-8. [10] Vannucci P, Giannoni B, Pagnini P. Treatment of horizontal semicircular canal benign paroxysmal positional vertigo. J Vestib Res 1997;7:1-6. [11] Nuti D, Agus G, Barbieri M-T, Passali D. The management of horizontal-canal paroxysmal positional vertigo. Acta Otolaryngol 1998; 118:455-60. [12] Boleas-Aguirre MS, PéRez N, Batuecas-Caletrei OA. Bedside therapeutic experiences with horizontal canal benign paroxysmal positional vertigo (cupulolithiasis). Acta Oto-Laryngologica 2009; 129:1217-21.
American Journal of Otolaryngology–Head and Neck Medicine and Surgery 34 (2013) 72 – 74 www.elsevier.com/locate/amjoto
Case report
Granddaughter's somersault treats cupulolithiasis of the horizontal semicircular canal Dirk Czesnik, MD⁎, David Liebetanz, MD Department of Clinical Neurophysiology, Medical School Göttingen, Georg-August-University of Göttingen, Robert-Koch-Str. 40, Göttingen, Germany Revised 22 June 2012
Abstract
We report on a 61-year-old woman with cupulolithiasis of the right horizontal semicircular canal, which is usually difficult to treat. The patient reported that several years ago, similar symptoms relieved completely after having performed several somersaults together with her granddaughter. This time, repetitive somersaults were again effective to treat her benign paroxysmal positional vertigo. Acceleration during a somersault may induce an intracanalicular force strong enough to detach otoconia debris from the cupula. Rolling may then promote their reentrance into the utricle. This case suggests that repetitive somersaults may be an alternative treatment of cupulolithiasis of the horizontal semicircular canal. © 2013 Elsevier Inc. All rights reserved.
1. Introduction Paroxysmal dizziness is commonly caused by benign paroxysmal positional vertigo (BPPV). In most cases of BPPV, the posterior semicircular canal is affected. To a lower extent (5%–10%), the horizontal semicircular canal is involved [1–3]. The BPPV of the horizontal semicircular canal (h-BPPV) typically occurs during turning of the head while lying in a supine position. The etiology of h-BPPV can be variable. In most cases, it is idiopathic, but it can also occur posttraumatically or be induced by positioning maneuvers to treat PBPPV [4]. Clinically, h-BPPV is characterized by a bidirectional horizontal nystagmus, which is triggered as the head of the supine patient is turned from side to side. There are 2 subtypes of h-BPPV: (i) the canalolithiasis is caused by fragments of otoconia that move freely in the horizontal semicircular canal. In case of canalolithiasis, nystagmus is geotropic (toward the undermost ear) and more intense after rotation of the head toward the affected side. It is caused by the movement of otoconial debris within the long arm of the
⁎ Corresponding author. Department of Clinical Neurophysiology, Medical School Göttingen, Robert-koch-Str. 40, Göttingen. Tel.: +49 551 396650; fax: +49 551 3913614. E-mail address: [email protected] (D. Czesnik). 0196-0709/$ – see front matter © 2013 Elsevier Inc. All rights reserved. http://dx.doi.org/10.1016/j.amjoto.2012.07.002
horizontal semicircular canal, which stimulates utriculopetal endolymph flow. The vertigo is triggered by circular acceleration of the horizontal semicircular canal (ii), if cupulolithiasis otoconia are attached to the cupula or are trapped in the proximal segment of the canal near to the cupula. As the head of the supine patient is turned slowly from side to side, the cupula is displaced, which induces asymmetric vestibular stimulation [5,6]. The vertigo typically occurs when the head changes position; circular acceleration does not trigger vertigo. The nystagmus during cupulolithiasis of the horizontal semicircular canal is pathognomonically apogeotropic (away from the undermost ear) and more intense after rotation of the head toward the unaffected ear. Interestingly, there is no effective maneuver available for the treatment of the cupulolithiasis of the horizontal semicircular canal, to date. Here, we suggest that somersaulting may be a more effective treatment maneuver, which relieves symptoms within a few days. 2. Case report The 61-year-old woman turned to the emergency hospitalization because of rotary dizziness, which suddenly occurred after turning in bed that morning. The symptoms were accompanied by nausea and emesis. Several years ago, the patient experienced similar symptoms, which did not
D. Czesnik, D. Liebetanz / American Journal of Otolaryngology–Head and Neck Medicine and Surgery 34 (2013) 72–74
respond to conventional treatment, but remarkably disappeared after having performed several somersaults together with her granddaughter. Preexisting conditions are otosclerosis (surgery in 2000 and 2003) and migraine. The migraine is treated with sumatriptane (100 mg) and mergentane (50 mg) if required. 2.1. Neurologic examination Clinical examination revealed an apogeotropic nystagmus when moving her head left and right while lying in a supine position (see video). The nystagmus was more intense when the head was rotated to the left side. There were no pathological findings in any other cranial nerve. Sensory and motor examination, tendon reflexes, gait, and coordination showed no signs of pathology. Physical examination of the heart, lungs, and abdomen were, accordingly, without pathological findings. The typical history and clinical findings led to the diagnosis of the cupulolithiasis of the h-BPPV with the right horizontal semicircular canal affected. Other reasons for the dizziness were excluded. Because somersault had been previously therapeutically effective, we advised her to somersault repetitively again. Interestingly, only a few somersaults during hospitalization were necessary to decrease the symptoms remarkably and reduce nystagmus. After hospital discharge, we advised repeating the somersaults at home daily. A 6-week
73
follow-up, examination showed no remaining symptoms and no positional nystagmus.
3. Discussion The horizontal semicircular canal (HSC) variant of BPPV is the second most common cause of BPPV. However, so far there is no effective maneuver to treat the cupulolithiasis variant of HSC-BPPV. The most difficult challenge is to detach the otoconia from the cupula. The Lempert or Barbecue maneuver [7] and the forced prolonged position (FPP) [8–11] are the most common maneuvers to treat canalolithiasis. They all aim to reposition of the otoconia out of the semicircular canal. Controlled studies demonstrated an effectiveness of reposition of 75% and 75% to 90% for the Barbecue and FPP maneuvers, respectively. However, the effectiveness of cupulolithiasis treatment by these maneuvers is much lower because they are not likely to detach the otoconia from the cupula [8,12]. Boleas-Aguirre and coworkers [12] reported that only the combination and repetition of FPP or Lempert over 2 weeks were able to increase the effectiveness. However, the suggested FPP for more than 12 hours daily for at least 2 weeks often led to a reduced treatment compliance. In our case, repetitive somersaults reduced the symptoms significantly and effectively within a few days. Acceleration
Fig. 1. Body rotation (A) and rotation of the horizontal semicircular canal during a somersault (B; lateral view). The gray line illustrates the position of horizontal semicircular canal. (C) The left picture illustrates how acceleration in the forward-facing direction during the somersault induce an intracanalicular force strong enough to detach otoconia debris from the cupula. The right picture demonstrates how ongoing rolling may move loose otoconia debris back into the utricle.
74
D. Czesnik, D. Liebetanz / American Journal of Otolaryngology–Head and Neck Medicine and Surgery 34 (2013) 72–74
in the forward-facing direction during the somersault may induce an intracanalicular force strong enough to detach otoconia debris from the cupula. The ongoing rolling may move loose otoconia debris back into the utricle (Fig. 1). This case suggests that repetitive somersaulting may be an alternative treatment of the HSC variant of BPPV. To evaluate the effectiveness, a prospective study in more patients is needed. Supplementary data to this article can be found online at http://dx.doi.org/10.1016/j.amjoto.2012.07.002. References [1] Pagnini P, Nuti D, Vannucchi P. Benign paroxysmal vertigo of the horizontal canal. ORL J Otorhinolaryngol Relat spec 1989;51:161-70. [2] McClure JA. Horizontal canal BPV. J Otolaryngol 1985;14:30-5. [3] Cakir BO, Ercan IC, Akir ZAC, Enol C, Sayin I, Turgut S. What is the incidence of horizontal semicircular canal benign paroxysmal positional vertigo? Otolaryngol Head Neck Surg 2006;134:451-4. [4] Baloh RW, Jacobson K, Honrubia V. Horizontal semicircular canal variant of benign positional nystagmus? Neurology 1993;43:2542-9.
[5] Fife TD. Recognition and management of horizontal canal benign positional vertigo. Am J Otol 1998;19:345-51. [6] Nuti D, Vannucchi P, Pagnini P. Benign paroxysmal positional vertigo of the horizontal canal: a form of canalolithiasis with variable clinical features. J Vestib Res 1996;6:173-84. [7] Lempert T, Tiel-Wilck K. A positional maneuver for treatment of horizontal canal benign positional vértigo. Laryngoscope 1996;106: 476-8. [8] Chiou WY, Lee HL, Tsai SC, Yu TH, Lee XX. A single therapy for all subtypes of horizontal canal positional vertigo. Laryngoscope 2005; 115:1432-5. [9] Cassani AP, Vannucci G, Fattori B, Berretini S. The treatment of horizontal canal positional vertigo: our experience in 66 cases. Laryngoscope 2002;112:172-8. [10] Vannucci P, Giannoni B, Pagnini P. Treatment of horizontal semicircular canal benign paroxysmal positional vertigo. J Vestib Res 1997;7:1-6. [11] Nuti D, Agus G, Barbieri M-T, Passali D. The management of horizontal-canal paroxysmal positional vertigo. Acta Otolaryngol 1998; 118:455-60. [12] Boleas-Aguirre MS, PéRez N, Batuecas-Caletrei OA. Bedside therapeutic experiences with horizontal canal benign paroxysmal positional vertigo (cupulolithiasis). Acta Oto-Laryngologica 2009; 129:1217-21.