We present a case of a true aneurysm of the superior labial artery. A 97-year-old woman visited our department complaining of swelling on the left side of the buccal region. A superior labial aneurysm was identified as the cause of buccal swelling by Doppler ultrasonography, enhanced computed tomography, and dynamic contrast-enhanced magnetic resonance imaging, and we performed surgical resection under general anesthesia. Histological and immunohistochemical examinations confirmed the diagnosis of a true aneurysm of the superior labial artery. The patient remains well 5 years and 3 months after operation. To the best of our knowledge, this is the first reported case of a true aneurysm of the superior labial artery in Japan.
