Aneurysms of the A1 segment of the anterior cerebral artery tend to develop in combination with various vascular anomalies of the A1 segment. Arterial branches that originate from the A1 segment and perfuse cortical regions are known to be rare. In this report, we describe a 48-year-old woman who presented with a ruptured aneurysm at the origin of an anomalous cortical artery arising from the A1 segment, for which microsurgical neck clipping was performed. Intraoperatively, the anomalous artery was seen to originate from the A1 segment, running into the interhemispheric fissure. An aneurysm was located at the bifurcation of the anomalous artery and the A1 segment. Postoperative angiography showed that the anomalous artery has branched into the fronto-orbital artery and the frontopolar artery and terminated as the anterior internal frontal arteries. We report a rare case of an aneurysm arising from an anomalous callosomarginal artery that arose from the A1 segment and perfused the cortical region. It is of significance to recognize that an aneurysm can develop at the origin of an anomalous artery that arises from the A1 segment.