Parkinsonism associated with dural arteriovenous fistula (DAVF) has been described rarely; however, isolated parkinsonism as the presenting symptom of DAVF has not been reported. Here, we describe a patient with DAVF showing reversible isolated parkinsonism after embolization, which was well correlated with perfusion status of basal ganglia, suggesting that a perfusion defect was responsible for the pathogenesis of the parkinsonism in our DAVF patient.