BACKGROUND: An aneurysm at the fenestrated basilar artery usually arises at the proximal end of the fenestration, with few exceptions. We failed to reveal any case of aneurysm at this site associated with multiple fenestrations of the vertebrobasilar system. CASE REPORT: This 62-year-old male was admitted to our hospital 1 day after a sudden onset of headache. CT scan showed subarachnoid hemorrhage, and angiogram revealed an aneurysm at the distal end of the fenestrated basilar artery in association with two other fenestrations of the vertebrobasilar system. The limb of the basilar artery fenestration was trapped tightly with one clip through the transcondylar approach. Postoperative course was uneventful, and postoperative angiogram showed complete disappearance of the aneurysm. CONCLUSION: An extremely rare case of an aneurysm at the distal end of the fenestrated basilar artery in association with multiple fenestrations of the vertebrobasilar system is reported. The coexistence of vertebral artery fenestration and the fenestration of the vertebrobasilar junction may give a clue to the mechanism of occurrence of basilar artery fenestration.
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