Double ophthalmic arteries originating from the internal carotid artery and persistent primitive trigeminal artery.

Purpose: Ophthalmic artery (OphA) embryogenesis is a complex process with various origins. We describe herein a case in which the OphA stemmed from the persistent primitive trigeminal artery (PPTA), which has never been reported.

Methods: A man in his 70s was admitted to our hospital following the sudden onset of altered consciousness. A computed tomography (CT) scan revealed a cerebellar hemorrhage, brainstem compression, and hydrocephalus. The patient underwent emergency craniotomy for hematoma evacuation and decompression. Digital subtraction angiography (DSA) was performed 14 days postoperatively to determine the source of the bleeding.

Results: DSA of the right vertebral artery revealed retrograde filling of the left PPTA, originating from the distal two-thirds of the basilar artery (BA) and extending to the cavernous segment of the left internal carotid artery (ICA) as well as a small artery extending from the intermediate part of the PPTA into the orbit and passing through the superior orbital fissure (SOF). DSA of the left common carotid artery showed the typical supracavernous origin of the OphA.

Conclusion: This is the first reported case of double OphAs originating from the ICA and PPTA. It is possible that the OphA traversing the SOF had an anastomosis with the primitive trigeminal artery (PTA) during the embryonic period. Persistence of only the BA side of the PTA results in the OphA originating from the BA, whereas persistence of only the ICA side results in the OphA originating from the meningohypophyseal trunk.