Adamantinoma is an extremely rare tumour originating from bone that can be divided into classical and osteofibrous dysplasia (OFD)-like adamantinoma. This low-grade malignancy almost exclusively occurs in long bones, and… Click to show full abstract
Adamantinoma is an extremely rare tumour originating from bone that can be divided into classical and osteofibrous dysplasia (OFD)-like adamantinoma. This low-grade malignancy almost exclusively occurs in long bones, and only few cases of mandibular adamantinoma have been reported. Here, we report the case of a 30-year-old female with a 2-year history of right mandible tenderness. Radiological examinations showed a lytic lesion involving the right mandible without a well-defined margin. Biopsy confirmed the diagnosis of adamantinoma. She underwent a segmental mandibulectomy and reconstruction with a fibula flap. The definitive diagnosis was OFD-like adamantinoma. However, the tumour recurred after 5 years. The residual mandible and fibula flap were widely involved. A total mandibulectomy was performed. Five years later, there is no evidence of recurrence or metastasis. We recommend that adamantinoma be treated by radical resection with clear margins, and long-term surveillance is necessary due to the likely high local recurrence rate and the potential for late tumour metastasis.
               
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