Aneurysms of the azygos anterior cerebral artery (AACA) associated with fenestration are exceptional. We reported such a case. A 61-year-old woman presented with subarachnoid hemorrhage. Computed tomography angiography and digital subtraction angiography showed an aneurysm at the distal end of the AACA with a fenestration at its beginning. Stenting-assisted coiling of the aneurysm was performed. The postoperative recovery was uneventful. Follow-up DSA showed complete coiling of the aneurysm. By this case report and review of the literature, we found that the coexistence of the aneurysm and fenestration on the AACA was exceptional. Endovascular treatment of saccular AACA aneurysms is an appealing option.
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