Buttock claudication is usually caused by proximal arterial obstruction in the aorta or the common iliac artery. We report an unusual case of buttock claudication caused by isolated stenosis of the superior gluteal artery diagnosed by angiography. Both physical examination and noninvasive vascular explorations had been unremarkable. Twenty-six months after undergoing treatment by percutaneous transluminal angioplasty, the patient has no symptoms. Buttock claudication related to unilateral stenosis of the superior gluteal artery as observed in this case can be successfully managed by percutaneous transluminal angioplasty.