We report two cases of Meige’s disease. One shows complete syndrome with lesions in red nucleus, thalam us and frontal white matter, and the other shows incomplete syndrome with lesions in cerebellar vermis and basal ganglia. Functional implications and their relationship with the pathophysiologic mechanisms of Meige’s disease are speculated. Clinical symptoms which resemble other involuntary movement disorders, site of lesions found by various anatomical studies, and good to fair responses to antipsychotics suggest the dysfunction of dopaminergic system-hyperactivity of dopaminergic system induced by yet unidentified mechanisms. The remarkably high prevalence of depression preceding or following the advent of involuntary hyperkinetic motor symptoms is also of interest
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