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Findings A 12-year-old female given a retinal arteriovenous malformation and sclerotic vessels associated with retinal ischemia on fluorescein angiography, along with an ipsilateral ophthalmic arteriovenous malformation on magnetized resonance imaging. An 11-year-old male given retinal vascular engorgement and tortuosity along with a central retinal vein occlusion and secondary neovascularization. Conclusions and Relevance Retinal ischemia in Wyburn-Mason syndrome is hetero