A 62-year-old male presented with ruptured anterior communicating artery (ACoA) aneurysm manifesting as severe headache associated with the rare combination of median artery of the corpus callosum (MACC) and accessory middle cerebral artery (MCA). Computed tomography demonstrated diffuse subarachnoid hemorrhage. Left carotid angiography demonstrated an anomalous vessel originating from the ACoA complex and passing forward in the interhemispheric fissure between the two companion A
2 segments. This vessel was identified as the MACC. Another anomalous vessel originated from the left A
1-A
2 segment and passed into the sylvian fissure. This vessel was identified as the accessory MCA. Left frontotemporal craniotomy was performed to clip the neck of the aneurysm. After identifying both A
1 and A
2 segments, accessory MCA, and the MACC, the aneurysm neck was occluded successfully. The ACoA complex is one of the most frequent sites of vascular anomalies. Preoperative and intraoperative care is required to identify the presence of anomalies of the ACoA complex prior to clip placement, to avoid accidental damage or clipping, which may result in severe neurological deficits.
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