Maxillofac Plast Reconstr Surg.  2023;45(1):17. 10.1186/s40902-023-00380-y.

Ameloblastic carcinoma of the mandible: a case report

Affiliations
  • 1Oral Cancer Center, Tokyo Dental College, 5‑11‑13 Sugano, Ichigawa, Chiba, Japan
  • 2Department of Plastic, Oral and Maxillofacial, Teiko University School of Medicine, 2‑11‑1 Kaga Itabashi‑ku, Tokyo, Japan
  • 3Department of Oral Pathobiological Science and Surgery, Tokyo Dental College, 2‑9‑18 Kandamisaki, Chiyoda‑ku, Tokyo, Japan
  • 4Department of Oral Oncology, Oral and Maxillofacial Surgery, Tokyo Dental College, 5‑11‑13 Sugano, Ichikawa, Chiba, Japan
  • 5Division of Clinical Laboratory, Ichikawa General Hospital, Tokyo Dental College, 5‑11‑13 Sugano, Ichikawa, Chiba, Japan
  • 6Department of Biology, Tokyo Dental College, 2‑9‑7 Kandasurugadai Chiyoda‑ku, Tokyo, Japan
  • 7Department of Oral and Maxillofacial Surgery, Tokyo Dental College, 2‑9‑18 Kandamisaki‑cho, Chiyoda‑ku, Tokyo, Japan

Abstract

Background
Ameloblastic carcinoma is a malignant form of ameloblastoma and a very rare odontogenic tumor. We report a case of ameloblastic carcinoma that occurred after removal of a right-sided mandibular dental implant.
Case presentation
A 72-year-old female patient visited her family dentist with a complaint of pain around a lower right implant placed 37 years previously. Although the dental implant was removed with the diagnosis of periimplantitis, the patient experienced dullness of sensation in the lower lip and was followed up by her dentist, but after no improvement. She was referred to a highly specialized institution where she was diagnosed with osteomyelitis and treated the patient with medication; however, there was no improvement. In addition, granulation was observed in the same area leading to a suspicion of malignancy, and the patient was referred to our oral cancer center. The diagnosis of squamous cell carcinoma was made after a biopsy at our hospital. Under general anesthesia, the patient underwent mandibulectomy, right-sided neck dissection, free flap reconstruction with an anterolateral thigh flap, immediate reconstruction with a metal plate, and tracheostomy. Histological analysis of the resected specimen on hematoxylin and eosin staining showed structures reminiscent of enamel pulp and squamous epithelium in the center of the tumor. The tumor cells were highly atypical, with nuclear staining, hypertrophy, irregular nuclear size, and irregular nuclear shape, all of which were suggestive of cancer. Immunohistochemical analysis showed that Ki-67 was expressed in more than 80% of the targeted area, and the final diagnosis was primary ameloblastic carcinoma.
Conclusion
After reconstructive flap transplantation, occlusion was re-established using a maxillofacial prosthesis. The patient remained disease-free at the 1-year 3-month follow-up.

Keyword

Ameloblastic carcinoma; Ameloblastoma; Malignant odontogenic tumor
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