Pulmonary arteriovenous fistula (PAVF) is a venous malformation that permits right to left shunting of blood, bypassing the pulmonary capillary bed. Often PAVFs are seen in association with hereditary conditions. On the other hand, isolated PAVFs are rare and asymptomatic. There have been few reports of isolated PAVF related complications. A patient was referred to us with dysarthria and diplopia and history of surgically-treated PAVF. Further evaluations revealed a stroke in thalamic region. We found an open PAVF in a case of thalamic stroke.