This paper presents a case of a de novo unruptured anterior communicating aneurysm associated with a persistent primitive trigeminal artery. The patient was a 65-year-old female with tinitis, for whom computed tomography with contrast medium indicated right dural enhancement. Dural arteriovernous malformation was suspected and angiography was thus conducted. A right persistent primitive trigeminal artery was demonstrated but no aneurysm could be found. Six years later, magnetic resonance angiography indicated an aneurysm on the anterior communicating artery. It was subsequently removed by surgery. A persistent primitive artery is frequently found to accompany aneurysm but de novo aneurysm associated with such an artery does not appear in the literature to date. The authors emphasize the importance of follow-up angiography in cases of persistent primitive artery not accompanied by an aneurysm.