ArticleSurgical neurology international2021
Oro-mandibular dystonia in pediatric moyamoya disease: Two cases report.
Article in Surgical neurology international, 2021. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 2 papers.
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2 citing papers in PubMed, 2 citations in OpenAlex.
- Late-onset, first-ever involuntary movement after successful surgical revascularization for pediatric moyamoya disease - Report of two cases.Surgical neurology international · 2023Article
- Dystonia as the Initial Manifestation of Moyamoya Disease: A Case Report and Literature Review.Case reports in neurologyArticle
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5 authors at 3 institutions in 2 countries.
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Abstract
backgroundIn this report, we describe rare two pediatric cases that developed oro-mandibular dystonia due to moyamoya disease. CASE DESCRIPTION: A 7-year-old boy presented with oro-mandibular dystonia and transient weakness of the left extremities, and was diagnosed as moyamoya disease. Another 7-year-old boy developed oro-mandibular dystonia alone and was diagnosed as moyamoya disease. In both, cerebral blood flow (CBF) was markedly decreased in the involved hemispheres, including the basal ganglia and cerebral cortex. They successfully underwent combined bypass surgery and experienced no further attacks of oromandibular dystonia during follow-up periods. CBF almost normalized through surgical collaterals through direct and indirect bypass.
conclusionWhen treating patients with oro-mandibular dystonia, moyamoya disease should be listed as one of the differential diseases. The underlying mechanism of oro-mandibular dystonia in moyamoya disease is still unclear, but persistent cerebral ischemia in the basal ganglia and/or parietal lobe may play a key role to induce this rare symptom.
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