Objective: The study reports a case of lateral orofacial dystonia in a multiple system atrophy (MSA) patient treated with levodopa, and discusses the clinical characteristics of levodopa-induced dyskinesia (LID) in MSA patients.

Background: dyskinesia in MSA patient is considered to be a rare phenomenon that clinicians are not well aware of it. In our clinical practice, it occurs from time to time, showing distinct characteristics.

Method:
Clinical data and video of a case of MSA patient were retrospectively analyzed and relevant literatures were reviewed.

Results: The patient was a 61-year-old male with a chronic onset and progressive course of disease for 3 years. The main manifestations were rigidity and bradykinesia of the left extremities, involuntary spasm of left face, and myoclonus of left fingers. The unilateral orofacial spasm occurred around an hour following levodopa administration, and completely released within 4 hours. It was presented as the twitch of the facial muscles and blepharospasm, and he could merely open his left eye during the process. Non-movement symptoms included: urinary frequency and urgency, REM sleep behavior disorder, and constipation. MRI showed atrophy of cerebellum, pons, right putamen and bilateral frontal parietal lobes[figure1]. 18F- FDG-PET showed that the glucose metabolism in the pontine and left cerebellum was moderately decreased, and that in right posterior putamen was severely decreased[figure2] . Taking together, the patient was clinical diagnosed as possible MSA-P with peak-dose LID.

Conclusion: There are many differences between LID in MSA patients and Parkinson’s disease, and the hypometabolism and atrophy of striatum in MSA could be the underlying mechanism.

References: [1] JOST W H, LINGOR P, TöNGES L, et al. Dyskinesia in multiple system atrophy and progressive supranuclear palsy [J]. J Neural Transm (Vienna), 2019, 126(7): 925-32.
[2] BOESCH S M, WENNING G K, RANSMAYR G, et al. Dystonia in multiple system atrophy [J]. J Neurol Neurosurg Psychiatry, 2002, 72(3): 300-3.
[3] MARSILI L, BOLOGNA M, KOJOVIC M, et al. Dystonia in atypical parkinsonian disorders [J]. Parkinsonism Relat Disord, 2019, 66(25-33.

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MDS Abstracts - https://www.mdsabstracts.org/abstract/levodopa-induced-unilateral-orofacial-dystonia-in-multiple-system-atrophy-case-report-and-video-presentation/