Angiokeratoma is a cutaneous vascular disorder that occurs at any sites as trunk,extremities, fingers and toes etc. Although solitary or multiple cutaneous lesions have been reported, oral lesions have been very care. A 72-year-old man who had an exophtic, essile mass with dark red, black colored which located on buccal mucosa, was treated with excisional biopsy. He had no specific systemic history except for the medication of allopurinol, for treatment of gout since 10 years. Final diagnosis was determined as angiokeratoma by evaluation of clinical and histopathological finding, and the lesion has not been recurred for two months by decrease of allopurinol. It has been emphasized that the relationship between oral mucosal disease and the complication of allopurinol. Allopurinol is widely used for gout treatment, which we will report a case on the allopurinol-associated angiokeratoma in the oral mucosa.