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We aimed to study the value of blood oxygen level–dependent functional magnetic resonance imaging (BOLD-fMRI) in assessing cerebral hemodynamic changes for moyamoya disease (MMD).We recruited 15 healthy volunteers, 15 patients with MMD without dyskinesia, and 30 patients with MMD who experienced paroxysmal limb dyskinesia. The BOLD-fMRI scans were obtained during grasping motions of the left or right hand. Hemodynamic response curves in the primary motor cortices were generated. Six response parameters including negative response time (Tnr), maximum signal intensity of negative response, time to peak, maximum peak arrival time, maximum signal intensity of positive response, and positive response time were measured.The hemodynamic response curve in the primary motor cortices of MMD patients showed extended Tnr, prolonged positive response time, and delayed time to peak than those of the controls. The response curve showed longer Tnr and maximum peak arrival time in the primary motor cortices on the affected side of the dyskinesia group.Blood oxygen level–dependent fMRI is an effective technique to assess hemodynamic changes in patients with MMD.