Stereotactic radiosurgery is a well-known treatment tool for arteriovenous malformations (AVMs). The method has high validity and minimal invasiveness, but late-onset problems involving tumor formation and vasculopathy induced by radiation have been reported. We present a rare case of a radiation-induced ruptured de novo aneurysm following Gamma Knife surgery (GKS) for an AVM. A 17-year-old, right-handed male underwent GKS for AVM at the left parietal lobe. After 3 years, a follow-up angiogram showed a residual AVM at the angular gyrus. Then, a 2nd GKS was performed for the residual lesion. Six years after the 1st GKS, the AVM disappeared on the angiogram. Seven years later, he suffered a sudden onset of headache. A left carotid angiogram revealed a ruptured aneurysm at the M2-M3 junction of the middle cerebral artery parietal branch. Coil embolization was performed, and the aneurysm was occluded. The patient was discharged without any neurologic deficits.

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