Spontaneous Regression Followed by Rupture of an Untreated Brain Arteriovenous Malformation

BACKGROUND: Brain arteriovenous malformations (AVMs) are dynamic lesions. Unlike the recruitment of additional vessels and the growth of the nidus over time, which are well reported in the literature, spontaneous regression is much less frequent. Only a handful of cases reporting recanalization of spontaneously regressed AVMs have been published. AVMs that undergo spontaneous regression typically share structural and historical features, including previous hemorrhage, a small nidus, superficial venous drainage, and a single draining vein. Structural features and hemodynamic changes may predispose brain AVMs to spontaneous regression, and angiogenic processes are hypothesized to contribute to recanalization and hemorrhage. CASE DESCRIPTION: We present the case of a 37-year-old female who had been diagnosed 12 years ago with an unruptured Spetzler-Martin grade 3 AVM in the left medial occipital lobe after a history of progressively worsening migraines. The AVM was monitored for 1 year, but the patient was lost to follow-up until 11 years later, when the AVM nidus was found to be significantly smaller in size. One month later, the patient presented with acute intraparenchymal hemorrhage due to rupture of the AVM, which was then surgically resected. CONCLUSIONS: Our case is the second reported case of rupture of a spontaneously regressed AVM and the first reported case of this outcome with no history of previous hemorrhage. Close monitoring of brain AVMs may be warranted for longer periods of time than previously recommended.