A rare case of childhood subarachnoid hemorrhage (SAH) is reported. A 5-yea
r-old boy presented with SAH. A cerebral angiogram showed obstruction of th
e first segment of the left anterior cerebral artery (Al). The Al was surgi
cally trapped to prevent rebleeding. Proliferation of myofibroblasts was re
vealed in the intima of the Al. Cerebral infarction appeared in the territo
ry of the left middle cerebral artery (MCA) on day 2 after onset of SAH, an
d the patient suffered a second SAH on day 26. The second angiogram showed
stenosis and a saccular aneurysm in the left MCA. A second surgery, neck cl
ipping of the aneurysm, was needed. The multiple arterial lesions and histo
logical findings indicated that the SAHs and the infarction were caused by
the dissecting aneurysm affected with fibromuscular dysplasia (FMD). Intrac
ranial FMD is mainly detected in children and causes cerebral ischemia and
bleeding. Surgical treatment of childhood SAH should be discretely chosen w
ith due consideration for FMD.