Owing to the rarity of publications describing ameloblastic carcinoma, little is known about this entity in pediatric patients. To our knowledge, malignant transformation from an odontogenic cyst into an ameloblastic carcinoma in a pediatric patient has not been documented to date. We present the case of a 14-year-old boy in whom a large osteolytic lesion associated with an impacted right maxillary third molar germ was fortuitously detected by orthopanoramic radiography. With a preoperative clinical-radiographic diagnosis of odontogenic cyst, the patient underwent surgical enucleation of the lesion. Histologic evaluation rendered a diagnosis of follicular cyst with a focal area of ameloblastic carcinoma. The literature addressing ameloblastic carcinoma is reviewed.
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