Ameloblastic carcinomas are rare malignant lesions with 3 mandibular pediatric cases reported in the literature. We present a case of ameloblastic carcinoma in situ in a 15-year-old male with a right mandibular cystic lesion on computed tomography. The incisional biopsy revealed plexiform ameloblastoma. Due to the infiltrating and aggressive nature of the tumor, the patient underwent hemimandibulectomy and immediate reconstruction with a vascularized osteocutaneous fibula free flap. The final pathology was read as ameloblastic carcinoma in situ. Given the rarity of this disease in the pediatric population, this case report may be a valuable addition to the current literature.
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