The trigeminal artery provides a transient connection between the developing anterior and posterior cerebral circulations in early embryonic life, but occasionally persists. Its persistence is often associated with hypoplasia of the vertebral arteries. We describe a case of brain stem infarction occurring in a young man with a persistent trigeminal artery. We suggest that persistence of the trigeminal artery is a poorly recognised risk factor for brain stem ischaemia. We briefly review the embryology and complications of persistent trigeminal arteries.