BackgroundThe ophthalmic artery is the first branch of the internal carotid artery. It arises from the supraclinoid segment of the internal carotid artery within the subarachnoid space and enters the orbit via the optic canal. However, due to complex embryogenesis, the ophthalmic artery can arise from different parts of the internal carotid artery or the distal branches of the external carotid artery. This is usually associated with a variation in the course of the ophthalmic artery through the superior orbital fissure instead of coursing through the optic canal. The ophthalmic artery and its branches vascularise the eyeball and its contents. Consequently, information about its morphologic variation is essential for treating clinical conditions such as central retinal artery occlusion, retinoblastoma chemoembolization, and ophthalmic artery aneurysm.Case presentationWe report on two cases of the ophthalmic artery arising from the middle meningeal artery in one adult (33-year-old Indian female) and one pediatric (2-year-old African male) South African patient examined by digital subtraction angiography. The patients were diagnosed with arteriovenous malformations and bilateral retinoblastoma, respectively.ConclusionsThe ophthalmic artery plays a vital role in vision generation. Thus, its anatomy is of clinical interest to neurosurgeons, ophthalmologists, and interventional radiologists.