Moyamoya Disease Manifesting as Paroxysmal Kinesigenic Dystonia: A Case Report and Literature Review

Abstract Background: Moyamoya disease (MMD) is an uncommon intracranial vasculopathy that typically presents with ischemic or hemorrhagic stroke. Dyskinesia is a rare manifestation of MMD. Case presentation: We report a patient with MMD presenting with paroxysmal kinesigenic dystonia (PKD). Magnetic resonance imaging (MRI) and Digital subtraction angiography (DSA) were performed to confirm the diagnosis. Electroencephalogram and cognition assessment were also applied. A MRI of the brain showed infraction. DSA showed stenosis of the bilateral internal carotid and middle cerebral artery with prominent basal collaterals, which was compatible with MMD. We also review the cases of Moyamoya-induced dyskinesia reported previously. Conclusions: PKD might be a rare manifestation of MMD. MMD should be considered in the differential diagnosis of PKD.