Moyamoya disease is a progressive steno-oclusive arteriopathy of the circle of Willis that manifests on cerebral angiography with a characteristic net of vessels at the base of the brain representing collateralization. Described initially in Japan where children present with cerebral ischemia and adults with hemorrhagic stroke, it was recognized to have a more benign course in the United States, mainly causing cerebral ischemic events in young adults. Medical treatment is not intended to stop the progression of the disease and surgery is not exempt of risks. We present a patient with early moyamoya disease in the United States in whom primary transluminal angioplasty, a less invasive procedure, was performed to treat the stenotic arteriopathy with good immediate and sustained clinical and angiographic results, without evidence of restenosis at the site of angioplasty 2 years later.