A 57-year-old man developed visual loss following craniofacial surgery for an inflammatory ethmoidal sinus mass. Surgery was preceded by endovascular occlusion of the ophthalmic artery distal to central retinal artery (CRA). Routine angiography obtained immediately after endovascular ophthalmic artery occlusion showed patency of the CRA. He complained of visual loss one day after craniofacial surgery (2 days after embolization). Repeat emergency angiography confirmed the patent CRA. Ophthalmic examination and fluorescein angiography showed that the visual loss was due to anterior ischemic optic neuropathy (AION). Preservation of the CRA is critical during ophthalmic artery embolization to avoid visual complications. Neurosurgeons should be aware of the possibility of AION as a complication of ophthalmic artery embolization.