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Stroke in young adults is uncommon and may require extensive evaluation to elucidate an underlying cause. A 21- year-old male professional golfer experienced left side weakness, dysarthria, headache during golfing. Magnetic resonance imaging (MRI) revealed broad ischemia on right basal ganglia, frontal and temporal lobes. Magnetic resonance angiography (MRA) and 4-vessel angiography revealed beading of right intracranial internal carotid and middle cerebral arteries which suggests fibromuscular dysplasia. We report a case of intracranial fibromuscular dysplasia without renal involvement resulting in cerebral infarction in young male professional golfer. (J Korean Acad Rehab Med 2009; 33: 644-647)