We report a rare case of a ruptured anterior spinal artery (ASA) aneurysm caused by bilateral vertebral artery (VA) occlusion. Case Presentations: A 78-year-old man suddenly developed severe headache and slight hemiparesis, and was admitted to our hospital. Computed tomography (CT) revealed subarachnoid hemorrhage, mainly in the posterior fossa. On emergency angiography, the right VA terminated at the origin of the posterior inferior cerebellar artery (PICA), and anastomoses between the PICA and the anterior inferior cerebellar artery (AICA) were observed, in addition to a saccular 3-mm aneurysm with bleb originating from the PICA-AICA anastomosis. Left vertebral arteriography demonstrated that the left VA was occluded segmentally at the V4 level and revealed a tortuous arterial network filling the distal VA. Based on the location of the bleeding, the right VA aneurysm was considered to have ruptured. After balloon test occlusion of the right VA, parent artery occlusion was performed without complications. The patient had no neurological changes immediately after surgery, but several hours later, he stopped breathing. Retrospective analysis revealed an ASA aneurysm, which was determined to be the bleeding source. Although conservative treatment was performed, he died the fourth day after onset without neurological improvement. Conclusion: In cases of subarachnoid hemorrhage associated with bilateral VA occlusion, an aneurysm formed by hemodynamic stress may be the source of hemorrhage. It is important to suspect aneurysms in the extracranial collaterals, such as the ASA, and intracranial collaterals such as the PICA-AICA anastomosis. Keywords▶ anterior spinal artery aneurysm, subarachnoid hemorrhage, PICA-AICA anastomotic artery, VA rete mirabile This work is licensed under a Creative Commons Attribution-NonCommercial-NoDerivatives International License.