Squamous cell carcinoma arising in a dentigerous cyst is a rare lesion with distinct histopathology and stringent criteria for diagnosis.(1,2) During the past century, <60 cases have been reported.(3) Of the previously documented cases, all have occurred in adults. We present the case of a squamous cell carcinoma arising in a dentigerous cyst in a 16-month-old girl. This case poses interesting questions regarding the pathophysiology of these tumors and highlights the importance of maintaining a high index of suspicion regardless of age when evaluating cystic lesions of the mandible and maxilla.