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The formation of the ophthalmic artery (OA) is a complex process with two different proposed embryological steps for its development. Several anatomical variants have been described. We present a very unusual origin of the ophthalmic artery from the basilar trunk, in a 45-year-old male with a history of pontine hemorrhagic stroke. MRI and CTA showed evidence of previous hemorrhage in the pons and several intracranial arterial dysplastic dilatations. DSA confirmed several fusiform dilatations of the basilar trunk. In the left ICA, no ophthalmic artery was seen arising from the carotid siphon. The left ophthalmic artery arises from the basilar trunk and runs lateral to the cavernous sinus through the middle cranial fossa, entering the left orbit at the superior orbital fissure. The patient was treated conservatively. Two main theories for this anomaly are known, one from Lasjaunias and the other from Paget. To our knowledge, this basilar origin of the OA has only been described three times in the literature. For its origin, we propose a partial persistence of the trigeminal artery together with a dominance of the dorsal ophthalmic artery.