Desmoplastic ameloblastoma is an unusual type of ameloblastoma with special histologic and radiographic features and anatomic distribution which differs from those in the classic type of ameloblastoma. The purpose of this article is to assist the dental community in better understanding of this variation of odontogenic tumor. We present a patient with desmoplastic and conventional variant of ameloblastoma (hybrid lesion) localized in the anterior portion of the mandible. The tumor was expansile and painless. The radiographic finding showed an ill-defined mixed (radiopaque/radiolucent) lesion between roots of the lower left canine and the lateral incisor. Marginal resection was performed. Follow-up examination one year later revealed no recurrence of the tumor. Desmoplastic variant of ameloblastoma tends to infiltrate between bone trabeculae. Curettage leaves islands of the tumor within bone, which later manifest as recurrences. Therefore, for big lesions en bloc excision is the most widely used treatment to avoid recurrence.