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Hemidystonia and Hemichoreoathetosis as an Initial Manifestation of Moyamoya Disease
Chul Hyoung Lyoo, MD;
Seung Hun Oh, MD;
Jin-Yang Joo, MD, PhD;
Tae-Sub Chung, MD, PhD;
Myung Sik Lee, MD, PhD
Arch Neurol. 2000;57:1510-1512.
Objective To describe hemidystonia and hemichoreoathetosis in an adult patient with moyamoya disease without a previous history of cerebrovascular accident.
Design Case report.
Setting Tertiary care center.
Patient A 22-year-old woman suddenly developed dystonic spasms in her left hand and left foot after a severe emotional stress. The dyskinesia gradually subsided over the next 4 months. Five months after the onset, she suddenly developed choreoathetoid movement in her right hand and right foot.
Main Outcome and Results The patient had both somatic and cortical sensory deficits in the right hand and right foot. Magnetic resonance imaging of the brain showed an infarction at the right putamen and lesions involving the right frontal lobe and the left frontotemporoparietal lobe. Magnetic resonance cerebral angiography showed severe stenoses of both internal carotid arteries at the supraclinoid portion and numerous collateral vessels, compatible with moyamoya disease. Single photon emission tomography of the brain showed hypoperfused areas at the right frontal and left frontotemporoparietal lobes. The choreoathetosis of the right limbs improved markedly, along with improvement of sensory deficits.
Conclusions To our knowledge, this is the first report of an adult patient presenting with hemidystonia and hemichoreoathetosis as the initial manifestations of moyamoya disease.
From the Department of Neurology (Drs Lyoo, Oh, and Lee), Neurosurgery (Dr Joo), and Radiology (Dr Chung), Yongdong Severance Hospital, Yonsei University College of Medicine, Seoul, South Korea.
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