Ameloblastomas are benign aggressive odontogentic tumors that exhibit insidious growth rates with attainment of extensive dimesions. Because ameloblastomas are not usually symptomatic until late in their clinical course, few are detected early. This article reports an atypical case of a small, painful ameloblastoma arising between the roots of the mandibular left canine and lateral incisor in a 66-year-old female. The patient underwent an en bloc resection of the mandible, and no recurrence was demonstrated on an 11-month recall examination. Histopathology revealed a "hybrid" ameloblastoma with a pronounced desmoplastic pattern and acanthomatous changes. Practitioners should be vigilant for diminutive radiolucent lesions of the jaws with poorly defined borders. Timely recognition and intervention of ameloblastomas may improve treatment outcomes.