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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Published online: December 06, 2020
Accepted: October 9, 2020
© 2020 Published by Elsevier Ltd on behalf of International Association of Oral and Maxillofacial Surgeons.