Research in Vestibular Science

Dural Arteriovenous Fistula Presenting as Acute Unilateral Vestibulopathy: Won Jeong Son, Jieun Roh, Eun Hye Oh, Jae-Hwan Choi; Res Vestib Sci. 2023;22(4):127-131. Published online December 15, 2023; DOI: https://doi.org/10.21790/rvs.2023.22.4.127

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Abstract PDF: Intracranial dural arteriovenous fistula (dAVF) is characterized by an abnormal connection between branches of arteries and veins in the dura mater. Clinical manifestations of dAVF vary depending on their location, feeder arterial supply, amount of shunting, and most importantly, their venous drainage pattern. Acute vertigo has been rarely reported as an initial presentation of dAVF due to venous congestion in the brainstem. We report a patient who presented with acute right vestibulopathy without any brainstem signs in dAVF involving the transversesigmoid sinus. The patient showed abnormal caloric response but normal head impulse in the affected ear. Without any treatment, the patient’s symptoms gradually improved with a normalization of right canal paresis. Follow-up cerebral angiography also revealed a spontaneous regression of the shunt flow and reduction of venous drainage at the right transverse-sigmoid sinus. Based on the results of vestibular function tests and cerebral angiography, acute vertigo in our patient may be ascribed to impaired reabsorption of endolymph by focal venous congestion.

Visual Fixation-Induced Hemi-Seesaw Nystagmus: Hyun Sung Kim, Eun Hye Oh, Jae-Hwan Choi; Res Vestib Sci. 2023;22(1):19-22. Published online March 13, 2023; DOI: https://doi.org/10.21790/rvs.2023.22.1.19

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Seesaw nystagmus (SSN) is characterized by conjugate torsional nystagmus with opposite vertical components in the two eyes. The waveform may be pendular or jerk (hemi-seesaw nystagmus, HSSN), in which the slow phase corresponds to one half-cycle and the quick phase to the other. Pendular SSN and HSSN have distinct clinical presentations and underlying causes. The pathophysiology of pendular SSN may be instability of visuovestibular interactions, while the underlying mechanism for HSSN may be related to the ocular tilt reaction or an imbalance in vestibular pathways. We report a patient with HSSN due to unilateral mesodiencephalic infarction that becomes apparent during visual fixation only.

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Midbrain lesion-induced disconjugate gaze: a unifying circuit mechanism of ocular alignment?
Maximilian U. Friedrich, Laurin Schappe, Sashank Prasad, Helen Friedrich, Michael D. Fox, Andreas Zwergal, David S. Zee, Klaus Faßbender, Klaus-Ulrich Dillmann
Journal of Neurology.2024;[Epub] CrossRef

The Head-Bending Test in Posterior Semicircular Canal Benign Paroxysmal Positional Vertigo: Sol-lim Choi, Hyun-Sung Kim, Jae-Hwan Choi, Eun Hye Oh; Res Vestib Sci. 2022;21(4):99-103. Published online December 15, 2022; DOI: https://doi.org/10.21790/rvs.2022.21.4.99

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Abstract PDF: Objectives
This study aimed to investigate clinical significance of a head-bending test in benign paroxysmal positional vertigo (BPPV) involving the posterior semicircular canal (PC-BPPV).
Methods
We retrospectively recruited 256 patients with unilateral PC-BPPV between January 2016 and December 2021, and assessed the clinical characteristics of patients showing head-bending nystagmus (HBN).
Results
Of 256 patients, 138 (53.9%) showed HBN. Most patients (n=136, 98.6%) had downbeat nystagmus with (n=38) or without (n=98) torsional component. The remaining two patients had pure upbeat and torsional nystagmus, respectively. The torsional component was directed to the contralesional side in all. Between patients with and without HBN, there were no significant differences in clinical characteristics such as age, lateralization, types of BPPV (canalolithiasis or cupulolithiasis), and success rate of repositioning maneuver.
Conclusions
Head-bending test may be useful in predicting the diagnosis and lateralization of PC-BPPV.

Dissociated Vertical-Torsional Nystagmus in Vestibular Nucleus Lesion: Hyun-Sung Kim, Eun Hye Oh, Jae-Hwan Choi; Res Vestib Sci. 2022;21(1):19-23. Published online March 15, 2022; DOI: https://doi.org/10.21790/rvs.2022.21.1.19

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Abstract PDF: Dissociated vertical-torsional nystagmus is a unique form of nystagmus characterized by conjugate torsional but disparate vertical components. It has been mainly reported in internuclear ophthalmoplegia or medial medullary lesion involving the medial longitudinal fasciculus (MLF). The patterns of the nystagmus may be explained by a disruption of vestibulo-ocular reflex pathways from vertical semicircular canal or utriculo-ocular reflex within the MLF, but it is debatable. We described a dissociated upbeat-torsional nystagmus in a patient with vestibular nucleus infarction without involvement of MLF.

Bilateral Sudden Hearing Loss Caused by Basilar Artery Dissection: A Case Report: Ho Byung Lee, Jieun Roh, Hyun Min Lee, Jae-Hwan Choi; Res Vestib Sci. 2021;20(4):151-155. Published online December 15, 2021; DOI: https://doi.org/10.21790/rvs.2021.20.4.151

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Abstract PDF: Bilateral sudden sensorineural hearing loss (SNHL) is rare, but a possible symptom of vertebrobasilar ischemia. A 69-year-old female patient with hypertension and atrial fibrillation presented with bilateral sudden hearing loss and vertigo without other neurological symptoms. On examination, she had left-beating horizontal nystagmus with positive head impulse on the left side. Pure tone audiometry revealed severe SNHL on both sides. Brain computed tomography angiography showed a dissection in the proximal portion of the basilar artery (BA) with occlusion of the mid-BA and bilateral anterior inferior cerebellar arteries (AICA), which confirmed on transfemoral cerebral angiography (TFCA). Left common carotid angiography demonstrated retrograde blood flow into the BA and right AICA via the left posterior communicating artery. During TFCA, her right hearing loss dramatically improved. Nine days later, follow-up TFCA showed an improvement of antegrade flow of the BA and AICA. We suggest that vertebrobasilar ischemia can be suspected in patients with bilateral sudden SNHL who present with risk factors for stroke.

Isolated Floccular Infarction with Impairment of High-Frequency Vestibulo-Ocular Reflex: A Case Report: Eun Hye Oh, Hyun-Sung Kim, Jae-Hwan Choi; Res Vestib Sci. 2021;20(4):147-150. Published online December 15, 2021; DOI: https://doi.org/10.21790/rvs.2021.20.4.147

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Abstract PDF: The flocculus plays a crucial role in control of eye movements. Based on animal experiment, it is suggested that the flocculus is important for governing vestibuleocular reflexes. In humans, an isolated floccular lesion is extremely rare. We report oculomotor abnormalities in a patient with unilateral infarction of the flocculus, and compare our results with those of previously reported patients with floccular lesion.

기저동맥 박리로 발생한 양측 돌발성난청 증례: Ho Byung Lee, Jieun Roh, Hyun Min Lee, Jae-Hwan Choi; Received November 19, 2021 Accepted December 3, 2021 Published online December 3, 2021; [Accepted]

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Abstract: Bilateral sudden sensorineural hearing loss (SNHL) is rare, but a possible symptom of vertebrobasilar ischemia. A 69-year-old woman with hypertension and atrial fibrillation presented with bilateral sudden hearing loss and vertigo without other neurological symptoms. On examination, she had left-beating horizontal nystagmus with positive head impulse on the left side. Pure tone audiometry revealed severe SNHL on both sides. Brain computed tomography angiography (CT angiography) showed a dissection in the proximal portion of the basilar artery (BA) with occlusion of the mid-BA and bilateral anterior inferior cerebellar arteries (AICA), which confirmed on transfemoral cerebral angiography(TFCA). Left common carotid angiography demonstrated retrograde blood flow into the BA and right AICA via the left posterior communicating artery. During TFCA, her right hearing loss dramatically improved. Nine days later, follow-up TFCA showed an improvement of antegrade flow of the BA and AICA. We suggest that vertebrobasilar ischemia can be suspected in patients with bilateral sudden SNHL who present with risk factors for stroke.

고주파 전정안구반사의 이상을 보인 단독 타래 경색: Eun hye Oh, Jae-Hwan Choi, Hyun-Sung Kim; Received November 8, 2021 Accepted November 22, 2021 Published online November 22, 2021; [Accepted]

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Abstract: The flocculus plays a crucial role in control of eye movements. Based on animal experiment, it is suggested that the flocculus is important for governing vestibule-ocular reflexes (VORs). In humans, an isolated floccular lesion is extremely rare. We report oculomotor abnormalities in a patient with unilateral infarction of the flocculus, and compare our results with those of previously reported patients with floccular lesion.

Update on Genetic Study of Vestibular Disorder: Jae-Hwan Choi; Res Vestib Sci. 2021;20(2):37-44. Published online June 14, 2021; DOI: https://doi.org/10.21790/rvs.2021.20.2.37

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Abstract PDF: The wide availability of next-generation sequencing has enabled a rapid progress in the discovery of genetic variants associated with many disorders. However, the contribution of genetic factors in vestibular disorders is largely unknown due to the low prevalence of familial disorders and the clinical diversity. A detailed clinical characterization of patients and a choice of proper genetic tests are required to identify the genetic contribution in vestibular disorders. In this review, we will introduce a genetic approach for vestibular disorders and update the evidences to support the genetic contribution in vestibular disorders.

A Case of Spinocerebellar Ataxia Type 28: Jae-Hwan Choi, Eun Hye Oh, Seo Young Choi, Kwang-Dong Choi; Res Vestib Sci. 2021;20(1):33-36. Published online March 11, 2021; DOI: https://doi.org/10.21790/rvs.2021.20.1.33

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Abstract PDF: Spinocerebellar ataxia type 28 (SCA 28) is characterized by young-adult onset, very slowly progressive gait and limb ataxia, dysarthria, nystagmus, ptosis, and ophthalmoplegia. It is caused by a heterozygous pathogenic mutation in the AFG3L2. So far, approximately 80 cases with genetically-confirmed SCA 28 have been reported in the literature. We report a patient with mild gait ataxia and dysarthria who carried a known pathogenic mutation in the AFG3L2. This is the first report of genetically-confirmed SCA 28 in Korea.

Cogan Syndrome Accompanied with Meningitis: Hyung-Keun Kim, Eun Hye Oh, Na-Yeon Jung, Jae-Hwan Choi; Res Vestib Sci. 2018;17(3):119-123. Published online September 18, 2018; DOI: https://doi.org/10.21790/rvs.2018.17.3.119

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Abstract PDF: Cogan syndrome is a rare inflammatory disease characterized by intraocular inflammation and vestibulo-auditory dysfunction. The exact etiology of Cogan syndrome is still unknown, but is currently thought to be an autoimmune disease. Cogan syndrome can be accompanied with various conditions including fever, arthritis, skin rash, aortitis, central or peripheral nerve system involvement, lymphadenopathy, splenomegaly and diarrhea. We report a case of Cogan syndrome accompanied with meningitis.

Cogan’s syndrome accompanied with meningitis: Hyung-Keun Kim, Eun Hye Oh, Na-Yeon Jung, Jae-Hwan Choi; Received July 17, 2018 Accepted August 17, 2018 Published online August 17, 2018; [Accepted]

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Abstract: Cogan’s syndrome is a rare inflammatory disease characterized by intraocular inflammation and vestibulo-auditory dysfunction. The exact etiology of Cogan’s syndrome is still unknown, but is currently thought to be an autoimmune disease. Cogan’s syndrome can be accompanied with various conditions including fever, arthritis, skin rash, aortitis, central or peripheral nerve system involvement, lymphadenopathy, splenomegaly, diarrhea. We report a case of Cogan’s syndrome accompanied with meningitis.

Opsoclonus-Myoclonus Syndrome Associated with Scrub Typhus: You Jin Choi, Seo Young Choi, Jae-Hwan Choi, Kwang-Dong Choi; Res Vestib Sci. 2017;16(1):34-37. Published online March 15, 2017; DOI: https://doi.org/10.21790/rvs.2017.16.1.34

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Abstract PDF

Scrub typhus is an infective acute febrile disorder caused by the intracellular parasite Orientia tsutsugamushi. Neurological manifestations of scrub typhus are meningoencephalitis, cerebellitis, transverse myelitis, papilledema, and cranial nerve palsy. However, opsoclonus-myoclonus syndrome associated with scrub typhus has been rarely reported. A 59-year-old man developed vertigo, nausea, vomiting, and imbalance following scrub typhus infection for eight days. Examination showed eschar at the axilla, decreased mentality, and opsoclonus- myoclonus syndrome. Video-oculography disclosed opsoclonus with an amplitude of 15°–20° and a frequency of 6–8 Hz. The serum antibody titers to Orientia tsutsugamushi were 1:5,120, and cerebrospinal fluid analysis revealed pleocytosis. Brain magnetic resonance imaging was normal. Neurological symptoms and signs completely improved by systemic steroid and antibiotics treatment. Various mechanisms including direct disseminating inflammation or indirect immune modulation may give rise to neurological complications following scrub typhus.

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Citations to this article as recorded by

Scrub typhus meningoencephalitis presenting as opsoclonus myoclonus syndrome: A video-based case
Laxmikant Ramkumarsingh Tomar, Dhrumil Jatinbhai Shah, Utkarsh Agarwal, Atul Gogia, Anshu Rohatgi, CS Agrawal
Tropical Doctor.2022; 52(1): 192. CrossRef
Neurological Manifestations of Scrub Typhus
Sagar Basu, Ambar Chakravarty
Current Neurology and Neuroscience Reports.2022; 22(8): 491. CrossRef
Neurological facets of scrub typhus: A comprehensive narrative review
Divyani Garg, Abi Manesh
Annals of Indian Academy of Neurology.2021; 24(6): 849. CrossRef

Opsoclonus-Myoclonus Syndrome Associated with Scrub Typhus: You Jin Choi, Seo Young Choi, Jae-Hwan Choi, Kwang-Dong Choi; Received November 1, 2016 Accepted January 13, 2017 Published online January 13, 2017; [Accepted]

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Abstract: Scrub typhus is an infective acute febrile disorder caused by the intracellular parasite Orientia tsutsugamushi. Neurological manifestations of scrub typhus are meningoencephalitis, cerebellitis, transverse myelitis, papilledema, and cranial nerve palsy. However, opsoclonus-myoclonus syndrome associated with scrub typhus has been rarely reported. A 59-year-old man developed vertigo, nausea, vomiting, and imbalance following scrub typhus infection for eight days. Examination showed eschar at the axilla, decreased mentality, and opsoclonus-myoclonus syndrome. Video-oculography disclosed opsoclonus with an amplitude of 15-20° and a frequency of 6-8Hz. The serum antibody titers to Orientia tsutsugamushi were 1:5,120, and CSF analysis revealed pleocytosis. Brain MRI was normal. Neurological symptoms and signs completely improved by systemic steroid and antibiotics treatment. Various mechanisms including direct disseminating inflammation or indirect immune modulation may give rise to neurological complications following scrub typhus.

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