The authors report the case of a 34-year-old woman who presented with increasing headaches. Neuroimaging revealed bilateral anomalous vessels arising at the level of each ophthalmic artery, coursing rostromedially to join the anterior communicating artery (ACA), which harbored an aneurysm. Intraoperatively, the authors identified an abnormal gyral segmentation of the frontoorbital region, with a median gyrus separated from the olfactory tracts on each side by the gyrus rectus. No interhemispheric fissure was observed in the exposed area. This is the first report in the literature of an abnormal gyral segmentation in association with an infraoptic course of an ACA. Recognition of this possible gyral abnormality in association with this vascular anomaly is relevant for surgical exposure and treatment of aneurysms by clip placement.