We describe a case of bilateral infraoptic origin of the anterior cerebral arteries associated with an anterior communicating artery (ACOM) aneurysm. Anatomical variations of the anterior cerebral artery (ACA) are common; however, bilateral infraoptic course of the anterior cerebral artery is extremely rare. Since an infraoptic course of the ACA is associated with ACOM aneurysm formation, an understanding of the cerebrovascular anatomy and embryology is important for appropriate management of the aneurysm.
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