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A dural arteriovenous fistula (DAVF) refers to an abnormal direct connection between an intracranial artery and a dural venous sinus. A DAVF presenting with parkinsonism is rare, and is therefore easily misdiagnosed. Therefore, early consideration of DAVF in the differential diagnosis of reversible parkinsonism is necessary.We present the case of a 51-year-old male with progressive parkinsonism.He was diagnosed as straight sinus occlusion. Imaging studies revealed a DAVF associated with cerebral hypoperfusion of the lenticular nuclei and frontal lobe white matter.Endovascular embolization was performed through his left occipital artery.Treatment resulted in marked clinical improvement that a major improvement of parkinsonism was observed concomitant with no evidence of early venous drainage of this patient.DAVF should always be considered as a potential cause of progressive parkinsonism on account of its potential reversibility. Our case suggests a concomitant role of basal ganglia degeneration and frontal white matter hypoperfusion in the pathology of parkinsonism due to DAVF. However, the precise pathophysiology remains to be investigated.