ABSTRACT Involuntary movement is an uncommon manifestation of moyamoya disease. We describe a 12-year-old boy who suffered from involuntary movement after the first surgery but it improved dramatically after the second surgery. Initially, the patient underwent an encephalo-duro-arterio-myosynangiosis on the right side and encephalo-duro-periostealsynangiosis on the bifrontal side, which ameliorated his motor weakness in his left upper extremity. However, involuntary movement appeared in his right upper extremity. Secondly, the patient underwent encephalo-duro-arterio-myo-periostealsynangiosis on the left side. Thereafter, the patient’s involuntary movement completely resolved. The involuntary movement might occur due to the unequal distribution of the blood flow in the basal ganglia.
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