• Yuanxiao Wei, Ju Chen, Cancan Lu, Yijing Jiang, Zhiqiang Liu, Wenzhao Zhang, and Liqun Fang.
• The Fourth Affiliated Hospital of Harbin Medical University, Harbin, China.
• Medicine (Baltimore). 2023 Jun 23; 102 (25): e34008e34008.

RationaleMultiple system atrophy (MSA) is a group of adult-onset sporadic neurodegenerative diseases, mainly classified as MSA-C and MSA-P types. Due to the diversity of clinical symptoms, diagnosis faces a significant challenge. In the present case, we report a patient with isolated vertigo as the first presentation and abnormalities of the oculomotor system as the characteristic manifestations.Case ConcernA 64-year-old male had dizziness for 1 year, aggravated for 4 months, with accompanying symptoms of unsteady walking. Physical examination revealed spontaneous nystagmus, abnormal ataxic movements, and a broad basal gait. Video nystagmography revealed saccade intrusions and macrosaccadic oscillations, and opsoclonus. Magnetic resonance imaging (MRI) was unremarkable early, and positron emission tomography-computed tomography (PET-CT) announced a reduction in the volume of the cerebellum and brainstem.DiagnosisThe diagnosis of the possibility of MSA type-C, peripheral neuropathy, hypertension, and lacunar cerebral infarction was performed.ConclusionAtypical early clinical presentation may lead to delays, and identifying the critical problem through the patient simple clinical status requires long-term clinical experience and various ancillary examination tools.Copyright © 2023 the Author(s). Published by Wolters Kluwer Health, Inc.

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