A 63-year-old man presented with severe headache and left side motor weakness after transient loss of consciousness is reported. His right internal carotid artery(ICA) was ligated without distal bypass 37 years ago due to arteriovenous malformation(AVM) rupture. Preoperative cerebral angiography demonstrated short right ICA proximal stump as a result of previous carotid ligation and large AVM nidus with feeders of right middle cerebral artery(MCA), which was fed from the left ICA, and right posterior cerebral artery. Complete obliteration of malformed vessels was performed via the right transsylvian transcortical approach. Postoperative cerebral angiography demonstrated complete removal of the AVM with preservation of normal vasculature. The postoperative course was uneventful. Considering multiple patch calcification of AVM nidus, the AVM may be recanalized and grew gradually for a long time after Hunterian ligation.