A case of peripheral ameloblastoma with histopathologic evidence of clear cell components in a 46-year-old man is reported. The tumor was located in the lower right premolar region, and was excised en bloc from the mesial side of the first premolar to the distal side of the second premolar, with the patient under general anesthesia. On examination 4 years later no sign of recurrence was found.Histopathologically, the tumor had an ameloblastomatous structure with clear cells consisting of ovoid, clear cytoplasm and pyknotic nuclei. The area containing clear cells show direct transition to, or intermingling of, typical follicular areas, and tumor nests invaded bone, which suggested that the tumor had an aggressive biological character.