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Introduction
Malignant odontogenic neoplasms account for a small percentage of all odontogenic tumors and may be classified as carcinomas, sarcomas or carcinosarcomas (1). Ameloblastic fibro-odontosarcoma (AFOS) is an extremely rare odontogenic sarcoma, composed of benign odontogenic epithelium, a malignant mesenchymal component, and dental hard tissue (1). AFOS may either arise as a de novo lesion of the jaws (2,3), or following the malignant transformation of a pre-existing ameloblastic fibro-odontoma (AFO) (4–6). AFOS usually occurs in the second and third decades of life, with a predilection for the mandible (2–6). Due to its rarity and the lack of clinicopathological information, the diagnosis of AFOS is challenging. We herein report a de novo case of AFOS of the left mandible in a 31-year-old female patient, exhibiting active epithelial proliferation, which is considered as an uncommon finding in AFOS. In addition, the clinicopathological characteristics, clinical management and prognosis were also discussed, combined with a review of the relevant literature.
Case report
In September 2006, a 31-year-old woman was referred to the West China Hospital of Stomatology (Chengdu, China) due to a 6-month history of a swelling in the left mandible. The patient complained that the mass had expanded over the last 2 months, with associated pain.
On extraoral evaluation, the patient's face was asymmetrical due to a sizeable swelling over the left mandible, accompanied by limitation in opening the mouth. Intraorally, an exophytic neoplasm was observed, extending from the left lower premolar to the ascending ramus of the left mandible, measuring ~8×6×4 cm3, with migration of teeth 43–44 and missing teeth 45–47. No evidence of regional lymph node or distant metastasis was detected. The panoramic radiographic examination revealed a sizeable multilocular radiolucent lesion in the left mandible. The neoplasm extended from the left canine to the ramus of the left mandible, with indistinct margins and local perforation of the cortical plate. In addition, irregular radiopaque foci were observed within the lesion (Fig. 1).
The initial preoperative diagnosis was ameloblastoma, with local invasion and the possibility of malignant transformation. A left hemimandibular resection was performed, with tumor-free margins. Grossly, the tumor was exophytic and fleshy, with considerable destruction and replacement of the left mandible. The cut surface was grayish brown, with scattered firm tissue and hemorrhagic areas...