To describe a case of persistent trigeminal artery variant (PTAV) and presumed ophthalmic artery (OA) simultaneously arising from the inferolateral trunk (ILT).

A 53-year-old woman with an initial episode of convulsion underwent cranial magnetic resonance imaging (MRI) and MR angiography (MRA) of the intracranial region for the evaluation of brain and vascular lesions. The MR machine was a 3-T scanner.

MRI revealed no pathological brain lesions. On MRA, the left anterior inferior cerebellar artery (AICA) arose from the cavernous segment of the internal carotid artery (ICA), indicative of the PTAV. The arising point was more distal than the usual point, and the presumed OA also arose simultaneously from the short common trunk, which was considered the ILT.

There are two types of PTA: lateral (usual) and medial (intrasellar) PTA. The lateral-type PTA and PTAV arise from the ICA of the distal precavernous–proximal cavernous segment and take a course similar to that of the posterior fossa. The medial type arises slightly more in the distal cavernous segment than in the lateral type. The OA rarely arises from the cavernous segment of the ICA and enters the orbit via the superior orbital fissure. The origin of this type of OA is considered to be the ILT. We herein report a case of a PTAV and presumed OA arising simultaneously from an ILT. No similar case has been reported in the relevant English language literature.