Wyburn-Mason syndrome is a unilateral, sporadic disorder characterized by arteriovenous malformations (AVMs) of the retina, face, and central nervous system. Clinically, these lesions often manifest as dilated, tortuous retinal vessels.1 We report a complication of Wyburn-Mason syndrome, peripheral retinal neovascularization, that has not yet been described to our knowledge. Additionally, we present an iatrogenic consequence consisting of extensive retinal and choroidal ischemia after intra-arterial embolization therapy for posterior intracranial AVM extension.