The variations of origin of ophthalmic artery are extremely rare. Here, we aimed to present a patient had a rare variation of the ophthalmic artery.

The patient had a history of Moyamoya disease. The imaging studies revealed ophthalmic artery origination from basillar artery. In addition, M1-segment fusiform aneurysm, vasculitis and, as a variation, middle meningeal artery originating from the ophthalmic artery were also detected. We presented this case report as it is the first case with ophthalmic artery originating from anterior inferior cerebellar artery.

In our case we reported, M1 segment fusiform aneurysm, vasculitis, and as a variation, middle meningeal artery originating from the ophthalmic artery and anterior inferior cerebellar artery as ophthalmic artery origin and hereby present it for inclusion into the literature. Ophthalmic artery anatomical abnormalities occupies so little place in existing literature when nominating rare variations. Ophtalmic artery originating from anterior inferior cerebellar artery firstly defined as an ophthalmic artery variation including its way-out branch structures.