Research in Vestibular Science

Case Report

Paraneoplastic Cerebellar Degeneration Presented as Acute Vertigo: Sung-Hee Kim; Res Vestib Sci. 2022;21(1):24-27. Published online March 15, 2022; DOI: https://doi.org/10.21790/rvs.2022.21.1.24

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Abstract PDF: Paraneoplastic cerebellar degeneration is a rare neurological manifestation of nonmetastatic malignancy. Its usual manifestation is subacute to chronic dizziness, gait ataxia, and dysarthria. There have been only a few cases of paraneoplastic cerebellar degeneration with acute presentation. This study describes a patient with paraneoplastic cerebellar degeneration, who presented acute vestibular syndrome and then episodically developed horizontal gaze-evoked nystagmus and gait ataxia.

Review

Understanding Vestibular Information Processing in Velocity-Storage Circuit and Application to Interpreting Clinical Manifestation of Vestibular Disorders: Jeong-Yoon Choi; Res Vestib Sci. 2021;20(3):81-92. Published online September 15, 2021; DOI: https://doi.org/10.21790/rvs.2021.20.3.81

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Abstract PDF: The velocity-storage circuit comprised of bilateral vestibular nucleus complexes, commissural fiber, and nodulus and uvula functions in refining the raw vestibular signal to estimate rotational velocity, gravity direction, and inertia. In this review, we pursued the functional significance of this velocity-storage circuit and how this physiologic knowledge could help us understand the clinical symptoms and signs of patients with vestibular disorders.

Case Reports

Neurotological Findings in a Patient with Glufosinate Ammonium Intoxication: Ji Young Kim, Joo Yeon Ham, Seong-Hae Jeong; Res Vestib Sci. 2020;19(4):138-140. Published online December 15, 2020; DOI: https://doi.org/10.21790/rvs.2020.19.4.138

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Abstract PDF Supplementary Material: The upward deviation could be explained by loss of inhibitory inputs from the cerebellum onto the brainstem anterior semicircular canal projections for upward vestibulo-ocular reflex, which would lead to an upward bias in static eye position. Therefore, upward gaze deviation has been reported in comatose patients after resuscitation and diffuse cerebrocerebellar damage sparing the brainstem. Herein, we report a patient with ingestion of glufosinate ammonium presented with cerebellar ataxia and ocular motor findings suggestive of cerebellum involvement such as upward gaze tendency, spontaneous downbeat, gaze-evoked nystagmus, perverted head impulse test, and impaired smooth pursuit.

Positional Hemiseesaw Nystagmus in Ataxia with Oculomotor Apraxia Type 2 due to a Novel Senataxin Gene Mutation: A New Phenotype: Sung-Hee Kim, Ja-Hyun Jang, Ji-Soo Kim; Res Vestib Sci. 2020;19(1):12-15. Published online March 15, 2020; DOI: https://doi.org/10.21790/rvs.2020.19.1.12

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Abstract PDF Supplementary Material

Ataxia with ocular motor apraxia type 2 (AOA2) is an autosomal recessive disorder that is characterized by adolescent-onset gait ataxia, peripheral neuropathy, ocular apraxia, and cerebellar atrophy. A 19-year-old male with AOA2 from a novel SETX mutation showed distinct oculomotor abnormalities that included spontaneous and gaze-induced downbeat nystagmus, impaired smooth pursuit, and reversed catch-up saccades during horizontal head impulse tests, as well as peripheral neuropathy involving the lower extremities and mild slowing of frontal processing. He also showed positional hemiseesaw nystagmus in the supine and straight head-hanging positions. Positional hemiseesaw nystagmus is a new manifestation of hereditary cerebellar ataxia and may be explained by a gravity-dependent position-induced error in estimating the tilt in the roll plane due to dysfunction of the tilt-estimator circuit.

Citations

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A Case of AOA2 With Compound Heterozygous SETX Mutations
Hee Jin Chang, Ryul Kim, Minchae Kim, Jangsup Moon, Man Jin Kim, Han-Joon Kim
Journal of Movement Disorders.2022; 15(2): 178. CrossRef

Rapidly Progressive Ophthalmoplegia and Brainstem-Cerebellar Dysfunction in Rhombencephalitis Caused by Listeria monocytogenes: Sung-Sik Kim, Jong-Hun Kim, Jin-Heui Lee, Seung-Han Lee; Res Vestib Sci. 2017;16(1):29-33. Published online March 15, 2017; DOI: https://doi.org/10.21790/rvs.2017.16.1.29

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Abstract PDF: Listeria monocytogenes (L. monocytogenes) is a rare, but important bacterial pathogen causing central nervous system infection in the elderly, pregnant women, and immunocompromised patients. A 60-year-old man with diabetes presented with headache, fever and rapidly progressive ophthalmoplegia. Based on serological and MRI studies, he was diagnosed with rhombencephalitis due to L. monocytogenes. The patient recovered without complications with urgent initiation of empirical antibiotics and the pathogen-specific antibiotic treatment. L. monocytogenes should be considered as a cause of rhombencephalitis presenting as external ophthalmoplegia.

Isolated Infarction of Anterior Cerebellar Vermis: Sung-Hee Kim, Ji-Soo Kim; Res Vestib Sci. 2016;15(4):147-150. Published online December 12, 2016; DOI: https://doi.org/10.21790/rvs.2016.15.4.147

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Abstract PDF

The anterior cerebellar vermis has been known to act in coordination of gait and postural adjustment of the trunk and legs. However, oculomotor abnormalities in an isolated anterior vermian lesion have not been described in the literature. A 59-year-old man presented with acute non-rotatory dizziness and disequilibrium. Neuro-ophthalmologic examination found impaired smooth pursuit and hypometric saccades in the contralesional direction, and disconjugate ipsiversive ocular torsion, but without spontaneous or gaze-evoked nystagmus. Imaging study showed an infarction restricted to the rostral end of right cerebellar vermis involving the lingual and central lobules. The anterior cerebellar vermis participates in the maintenance of axial posture and gait, and also in the control of ocular motor and vestibular systems.

Citations

Citations to this article as recorded by

Free water imaging unravels unique patterns of longitudinal structural brain changes in Parkinson’s disease subtypes
Abigail E. Bower, Sophia J. Crisomia, Jae Woo Chung, Justin P. Martello, Roxana G. Burciu
Frontiers in Neurology.2023;[Epub] CrossRef

Symposium I

Ataxia and Diplopia: Jae-Deuk Seo, In Soo Moon, Kwang-Dong Choi; Res Vestib Sci. 2013;12:S31-S40. Published online June 1, 2013

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Abstract PDF: Ataxia is a neurological sign consisting of lack of voluntary coordination of muscle movements, which may result from abnormalities in different parts of the nervous system including the cerebellum and its connecting pathway, proprioceptive sensory pathway, and vestibular system. In the diagnosis of disorders characterized by cerebellar ataxia, the mode of onset, rate of development, and degree of permanence of the ataxia are of particular importance. In adults, paraneoplastic and demyelinating cases account for the largest proportion of subacute onset, and hereditary forms are the usual cause of very slowly progressive and chronic ones. The last category of genetic ataxias now constitute a large and heterogeneous group for which the basis has been established in most cases. Misalignment of the visual axes-strabismus-causes the two images of a seen object to fall on noncorresponding areas of the two retinas, which usually causes diplopia, the sensation of seeing an object at two different locations in space. Accurate diagnosis of diplopia and strabismus require detailed history on the symptomatology and ocular motor examination consisting of assessment of the range of eye movements, subjective diplopia testing, and cover and alternate cover tests.

Case Reports

Periodic Alternating Nystagmus in Patients with Cerebellar Abscess: Hyung Lee, Hyun Ah Kim; Res Vestib Sci. 2016;15(1):27-29.

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Abstract PDF: Periodic alternating nystagmus (PAN) is characterized by a periodical reversal in the direction of the nystagmus. Acquired PAN is caused by lesions of the inferior cerebellar vermis, causing disinhibition of the velocity storage mechanism, which is mediated by the vestibular nuclei. An eighty-year-old woman with abscess in midline cerebellum experienced dizziness and imbalance. We observed short period PAN with 7?8 seconds.

Two Cases of Central Vertigo Presenting as Apogeotropic Direction Changing Positional Nystagmus: Min Chul Park, Jin Su Park, Min Beom Kim, Jae Ho Ban; Res Vestib Sci. 2014;13(2):57-62.

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Abstract PDF: Positional vertigo and nystagmus without focal neurological symptoms and signs are characteristic features of benign paroxysmal positional vertigo (BPPV). And the apogeotropic positional nystagmus can be diagnosed as cupulolithiasis of the horizontal semicircular canal. However, cerebellar lesion involving especially nodulus could be initially presented as positional vertigo like a BPPV without other neurologic signs. In most of the patients with cerebellar involvement, initial presentation shows dysarthria, ataxia, headache, nausea, vomiting and unsteadiness. But in some central lesions, positional nystagmus might be observed in head roll test as if BPPV was presented. It is very important for clinicians of dizziness care unit to differentiate central positional vertigo (CPV) from BPPV. But it is difficult to diagnose CPV at initial visit by history and physical exam only. Therefore, we introduce two cases with cerebellar infarction and hemorrhage initially presenting isolated positional vertigo mimicking BPPV.

Original Article

Initiation of Smooth Pursuit in Acute Cerebellar Infarction: A Preliminary Study: Tae Ho Yang, Sun Young Oh; Res Vestib Sci. 2013;12(2):47-53.

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Abstract: Background and Objectives: The cerebellar lesion causes an initiation deficit of smooth-pursuit eye movement depending on the location of the lesion. We investigated the initiation of smooth pursuit in patients with cerebellar infarction and in healthy subjects, using step-ramp stimuli. Materials and Methods: Ten patients with cerebellar infarction documented by brain magnetic resonance imaging and fifty healthy subjects are recruited. To estimate the initiation of smooth pursuit, the onset latency and initial acceleration during the first 100ms of the horizontal smooth pursuit were estimated using the step-ramp target stimuli (5°/sec, 10°/sec, and 20°/sec). Results: In healthy subjects, onset latency of pursuit was shortened and initial acceleration was increased as target velocity was increasing. In patients with unilateral cerebellar infarction, the onset latency of ipsilesional smooth pursuit was significantly delayed at the target velocities of 10°/sec and 20°/sec. For the fast target velocity of 20°/sec, there was significant decrease of the initial acceleration of contralesional pursuit. Conclusion: In comparison with the healthy subjects, the patients with unilateral cerebellar lesions showed significant delay of pursuit onset and decrease of initial eye acceleration in the fast target velocity. These results support that the cerebellar lesions affect not only steady-state smooth pursuit gain but also the processing time required to initiate smooth pursuit, i.e., onset latency and initial acceleration. More extensive study is needed to confirm the role of cerebellum for parametric adjustment of each component of smooth pursuit.

Case Report

A Case of Cerebellar Hemangioma Mimicking Peripheral Vestibulopathy: Jae Ho Ban, Nam Hoon Lee, Hyun Jin Choi, You Sam Won; J Korean Bal Soc. 2008;7(1):81-84.

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Abstract PDF: Central vertigo of cerebellar origin may present the syndromes similar to those of peripheral vertigo such as vestibular neuronitis. The character of those vertigo syndrome depend on the location, extent, and the etiology of the lesion such as stroke, inflammation, mass. Cavernous hemangioma may be clinically silent, but can cause variable neurologic manifestations including central vertigo if it affects the vestibular system. We report a rare case of cerebellar cavernous hemangioma with the vertigo syndrome closely mimicked vestibular neuronitis.

Original Articles

Tilt Suppression of the Post-rotatory Nystagmus in Cerebellar Nodular Lesions: Sun Young Oh, Kwang Dong Choi, Jung Eun Kim, Ja Won Koo, Ji Soo Kim; J Korean Bal Soc. 2007;6(2):161-166.

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Abstract PDF: Background and Objectives: Head tilt at the end of step rotation about a vertical axis decreases the time constant (TC) of the post-rotatory nystagmus, which is known as tilt-suppression of the vestibulo-ocular reflex (VOR). Tilt suppression of the VOR is mediated by the cerebellar nodulus and ventral uvula and is eliminated after surgical ablation of those structures. However, studies on the tilt suppression of the VOR have been sparse in humans with cerebellar lesions. Materials and Methods: Five patients with circumscribed cerebellar lesions involving the nodulusor ventral uvula underwent recording of spontaneous and positional nystagmus, and the VOR. To evaluate tilt suppression of the VOR, the participants pitched their head forward at the end of step rotation about a vertical axis both in the clockwise and counter-clockwise directions. Results: The VOR gain was increased in a patient with infarction in the territory of the medial posterior inferior cerebellar artery while the gain of visually enhanced VOR was normal in all the patients. The time constants of perand post-rotatory nystagmus was increased in a patient with increased VOR gain and the tilt suppression of the post-rotatory nystagmus was impaired in two patients, either uni- or bilaterally. Spontaneous downbeat and central positional nystagmus were frequently accompanied. Conclusions: Nodular lesion may impair tilt suppression of the VOR. Measurement of tilt suppressive effect of the VOR may provide a valuable tool for evaluating the nodular dysfunction.

Clinical Manifestations of Cerebellar Infarction Mimicking Unilateral Vestibulopathy: Seung Suk Lee, Jae Ho Ban, Chee Yeul Park, No Hee Lee, Jong Kyu Lee; J Korean Bal Soc. 2006;5(2):229-234.

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Abstract PDF: Background
and Objectives: Pseudo-labyrinthine symptom without any other accompanying neurologic symptoms or signs occur with cerebellar infarction. The prognosis and management of cerebellar infarction differ from those of vertigo associated with unilateral vestibulopathy. The objective of study was to analyze the clinical symptoms of cerebellar infarction mimicking unilateral vestibulopathy according to the infarction territory. Materials and Method: Among 273 patients who showed pseudo-labyrinthine symptoms or signs, 28 patients were diagnosed cerebellar infarction on MRI between January 2003 and October 2006. Out of the 28 patients, 9 patients accompanying with focal neurologic symptoms or signs in early stage were excluded, and a retrospective analysis of total 19 patients was done through chart reviews. Using diffusion-weighted imaging, we divided 19 patients into two groups, AICA and PICA territory infarction. Clinincal features, outcome of audiologic and vestibular function test were compared between the groups.
Results
Among the 19 subjects, 10 were males and 9 were females, the mean age was 63.1±4.0. On MRI, 5 (26.3%) subjects showed infarction in unilateral AICA territory, 14(73.7%) in unilateral PICA territory. In the patients with AICA territory infarction, audiological tests confirmed unilateral sensorineuronal hearing loss in all 5 subjects. On V-ENG, 4/5 (80%) demonstrated horizontal or mixed horizontal torsional spontaneous nystagmus, and 4/5 (80%) had a canal paresis to caloric stimulation. In several days of hospital stay, 3/5 (60%) showed new neurologic signs, facial numbness. In the patients with PICA infarction, there was no hearing change. On V-ENG, 12/14(85.7%) demonstrated horizontal or mixed horizontal torsional spontaneous nystagmus, and 7/14 (50%) had a canal paresis to caloric stimulation. In several days of hospital stay, 10/14 (71.4%) accompanied with cerebellar ataxia.
Conclusion
Cerebellar infarction simulating unilateral vestibulopathy is more common than previously thought. Early recognition of the cerebellar infarction showing pseudo-vestibular symptom may allow specific management. Also taking into account that clinical features differ by infarction territory will assist in determining the patient’s status of the disease. Key Words : Cerebellum, Infarction

A Promotive Effect of Low Level Laser on Hair Cell Viability in Postnatal Organotypic Culture of Rat Utricles: Sun Young Oh, Kwang Dong Choi, Jae Moon Kim, Jei Kim, Seong Ho Park, Ji Soo Kim; J Korean Bal Soc. 2006;5(1):35-43.

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Abstract PDF: Background
and Objectives: To culture and maintain mammalian hair cells is still a big challenge. In this study, long-term organotypic culture of rat utricular maculae was established to study vestibular hair cell. The effects of low level laser on hair cell viability in postnatal organotypic culture of rat utricles were investigated. Materials and Method: Uticular explants were prepared from postnatal 2 to 7 rats and cultured. To improve hair cell survival, the utricles were irradiated daily with low level laser. Whole-mount utricles were stained with FM1-43 which is known to be an efficient marker to identify live hair cells in cultured tissues. Such cells visualized directly through tissue culture dish with cover glass bottom by Confocal laser scanning microscope at specific time points.
Results
The explanted utricular hair cells were cultured for up to 31 days in in vitro culture system. In low level laser irradiation group, utricular hair cells were more survived at 24 DIV and 31 DIV.
Conclusion
These results suggest that low level laser promotes hair cell viability in utricular explants. Key Words : Organotypic culture, Low level laser, FM 1-43, Utricle

Review

Central Positional Nystagmus from Focal Brain Lesion: Ja Won Koo, Kwang Dong Choi, So Young Moon, Seong Ho Park, Ji Soo Kim; J Korean Bal Soc. 2004;3(1):129-135.

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Abstract PDF: Department of Otolaryngology, Head and Neck Surgery1, and Neurology2, Seoul National University Bundang Hospital, Seoul National University College of Medicine, Seongnam, Korea Background and Objectives : Central positional nystagmus is induced by positional changes from brainstem or cerebellar lesions. Differentiation central from peripheral positional nystagmus is important in clinical practice. To delineate characteristics of the positional nystagmus observed in central lesions, we analyzed the parameters of positional nystagmus from focal brain lesions. Materials and Methods : Ten patients with central positional nystagmus were recruited from the dizziness clinic of Seoul National University Bundang Hospital. All the patients had focal brainstem or cerebellar lesions documented by magnetic resonance imaging. The nystagmus was observed with and without fixation by using Frenzel glasses or Video Goggles. The nystagmus was videotaped or recorded with video-oculography. Provoking positional maneuvers, direction, latency, duration, phase reversal, and fatigue phenomenon of the nystagmus were analyzed.
Results
: Of the 10 patients, seven had infarctions in the lateral medulla or inferior cerebellum while two experienced cerebellar hemorrhage and remaining one showed a compression of the ventrolateral medulla by cavernous malformation of the vertebral artery. The directions of the positional nystagmus were variable depending on the lesions and provoking maneuvers. Most patients exhibited direction-changing nystagmus without latency, direction-reversal and fatigue phenomenon. However, some of the patients also showed patterns of nystagmus characteristic of peripheral positional nystagmus. In two of the four patients with infarction in the territory of medial posterior inferior cerebellar artery, the positional nystagmus was the only abnormal findings.
Conclusions
: Central positional nystagmus may share many characteristics with peripheral type of positional nystagmus. In individual cases, the patterns of nystagmus should be interpreted with caution in differentiating central from peripheral positional nystagmus. Considering the isolated positional nystagmus in some patients with caudal cerebellar lesions, systematic positional maneuvers should be applied to all the patients with vertigo Key Words : Positional nystagmus, Medulla, Cerebellum

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