Objective: Patients with moyamoya disease frequently develop ischemic and/or hemorrhagic events. A small number of patients with moyamoya disease, most of which are children or young adults, generate involuntary movements as an initial manifestation. However, the characteristics and clinical features are less known. In this case report, we present a geriatric case of moyamoya disease presenting with hemiballismus as a primary symptom, and a favorable clinical course achieved with oral

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... l (D2 receptor antagonist). Case presentation: A 79-year-old female developed unvoluntary choreoathetoid movement in the right upper and lower extremities. The gradually worsening symptom was accompanied with ballismus in two months. Head MR showed bilateral stenosis and occlusion of the terminal portions of the internal carotid arteries without any definitive destructive brain lesions. Abnormal vascular network at the base of the brain, so called moyamoya vessels, was confirmed in cerebral angiography. Moyamoya disease was diagnosed with these typical findings. After administration of haloperidol, the patient's involuntary movement gradually disappeared. Conclusion: Even though geriatric moyamoya with involuntary movement as an initial manifestation is extremely rare, meticulous evaluation including magnetic resonance (MR) is needed. We concluded that pharmacotherapy with a D2 receptor antagonist was adequate for symptom improvement, especially for the geriatric case that is not suitable for invasive vascular reconstructive surgery.