A 51-year-old man with preexistent lacunar infarctions in the basal ganglia and pons developed complex involuntary movements of his right arm 1 day after a purely cortical-subcortical ischemic infarction in the territory of the left middle cerebral artery sparing the basal ganglia. The movements were stereotyped and repetitive, affecting hand and fingers or proximal arm muscles alternatively or together. Although hyperkinesia after basal ganglia lesion has been described many times in the literature, it has not yet been reported after cortical-subcortical ischemic infarctions sparing the basal ganglia.