Abstract
A persistent primitive hypoglossal artery (PPHA) is a rare anomalous remnant that can be associated with vascular anomalies such as cerebral aneurysm, moyamoya disease, or arteriovenous malformation. An aneurysm developing in a PPHA associated with unilateral moyamoya disease has not previously been reported. We describe the case of a patient in whom an aneurysm ruptured on a PPHA associated with unilateral moyamoya disease. A 42-year-old man who developed a sudden severe headache followed by unconsciousness was diagnosed with a subarachnoid hemorrhage (SAH) at our hospital. Three-dimensional computed tomography (3D-CT) angiography and catheter cerebral angiography revealed an aneurysm on a left PPHA and left middle cerebral artery occlusion with basal moyamoya vessels. The aneurysm was successfully clipped. The postoperative course was uneventful, and the patient returned to work without neurological deficits three weeks after the surgery.