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.
METHODS: 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 peri-implantitis, 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.
CONCLUSIONS: 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.

Odontogenic carcinosarcoma (OCS) is a rare malignant odontogenic tumor (OT) with only a few cases reported in the literature. Its synonyms are ameloblastic carcinosarcoma, malignant mixed OT. It is characterized by a true mixed tumor showing malignant cytology of both epithelial and mesenchymal components. The tumor invaded into adjacent tissues by destroying the bone. A 24-year-old patient visited the outpatient clinic of GITAM Dental College and Hospital, with a chief complaint of growth in the lower right back tooth region for 6 months. Based on clinical and radiographic features, it has been diagnosed as an aggressive central jaw lesion. The patient was further referred for histological examination for confirmatory diagnosis. It has been diagnosed as an adenomatoid OT. The OCS most commonly affects the posterior part of the mandible. A larger number of cases were reported recently, and prolonged follow-up is needed to further clarify the nature of OCS.

Clear cell odontogenic carcinoma (CCOC) is a rare and aggressive malignant epithelial neoplasm, which occurs most frequently in the mandible of elderly patients. Morphologically, CCOC shares similar characteristics with other clear cell tumors, especially hyalinizing clear cell carcinoma of the salivary glands (HCCC). Both CCOC and HCCC are known to harbor EWSR1 rearrangements, especially the EWSR1-ATF1 gene fusion, which indicates a possible link between the two lesions. So far, this fusion has been demonstrated in five cases of CCOC in the literature. Herein, we add another CCOC case to the literature, which arose in the mandible of an 82-year-old female patient and was proven to harbor the EWSR1-ATF1 gene fusion. Immunohistochemically, this case was focally positive for CK7, CK14, CK19 and p63. The patient was referred to surgical treatment; however, she died of disease 2 months after the diagnosis, thereby demonstrating the aggressive nature of this tumor.

Ameloblastic Carcinoma is a rare malignant Odontogenic tumour with characteristic histopathology and clinical features which requires aggressive surgical treatment and surveillance and therefore differs from ameloblastoma. It is possible that ameloblastoma shows a variety of histologic and biologic behaviour ranging from benign to frank malignancy. Cases of ameloblastoma should thus be studied carefully, correlating their histologic pattern with biologic behaviour to direct subtle changes in histology that may predict the aggressiveness of the tumor. Thus the identifying features of Ameloblastic Carcinoma must be carefully known and recognized by dental professionals. The purpose of this article is to report a rare case of Ameloblastic Carcinoma and to highlight the clinical, radiological and variable histological features with possible differential diagnosis.