Moyamoya disease is a progressive, occlusive, cerebrovascular arteriopathy, characterized by bilateral stenosis of the distal internal carotid arteries or branches, with the development of compensatory collateral vessels. It is infrequently reported during infancy. We describe a 2-month-old boy presenting with stroke secondary to moyamoya disease, successfully treated with revascularization surgery at age 3 months. To our knowledge, this report represents the youngest such patient. This case and a review of the literature regarding moyamoya disease during infancy suggest that these patients are at significant risk for further ischemic events and respond well to prompt revascularization surgery.
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