A 40-year-old man suffered from a repeatedly recurrent desmoplastic ameloblastoma in the right maxillary anterior and premolar regions. During the first visit, the patient was provisionally histopathologically diagnosed with a developmental cyst, and it was confirmed to be unicystic ameloblastoma and resected. Four years later, the lesion recurred, and was diagnosed as a desmoplastic type of ameloblastoma and removed again. Then, 5 years after the second surgery, the lesion recurred again, and was diagnosed as a type containing a follicular pattern, recurrent ameloblastoma. A panoramic radiograph showed a multilocular and mixed radiolucent/radiopaque expansile lesion at the first visit, a unilocular cystic lesion confined to the premolar area at the second visit, and a small soap bubble appearance in the molar area in the final visit. Cone-beam computed tomographic images of the final recurrence of the tumor revealed multiple small cyst-like structures in the right maxillary anterior and posterior regions.