Arteriovenous malformations (AVM) of the brain are uncommon congenital lesions with a complex array of arterial to venous shunts. The common clinical manifestations include hemorrhage, seizures, headache and congestive heart failure. We recently saw a child who presented with bilateral ptosis, a rather rare and unusual manifestation of AVM. We discuss the reported ocular manifestations of AVM and the possible pathogenesis of the ptosis in this child.