Bilateral persistent primitive hypoglossal arteries (PPHA) associated with multiple aneurysms in a 59-year-old man are reported in this paper. Angiography of the left carotid artery revealed a PPHA on this side with a large aneurysm located at the junction of the basilar artery. This PPHA arose from the left internal carotid artery at the level of the C-2 vertebra. Right retrograde vertebral arteriography showed a PPHA and a small aneurysm at the trifurcation of the right middle cerebral artery. The necks of these aneurysms were successfully clipped. This seems to be the first case of bilateral PPHA associated with multiple aneurysms.