Background: Cerebral arteriovenous malformations (AVMs) are congenital lesions which rarely recur after its complete microsurgical excision. Case Description: This case report presents a 35 year-old woman who had referred with hemorrhagic AVM five years ago. This patient has recently undergone microsurgical excision for her left parieto-occipital Grade II AVM and surgical clipping of left posterior inferior cerebellar artery (PICA) aneurysm which concomitantly detected. She is the oldest case up to date whose AVM recurred as Grade III which is bigger than her first AVM associated with an aneurysm. Conclusion: Although some authors do not propose routine additional follow-up in adults after the initial negative postoperative angiogram, we believe that such patients should be followed clinically and radiolocically.