Abstract
BackgroundThe ameloblastic carcinoma is a rare malignant odontogenic tumor which rather occurs in the mandible than in the maxilla. Its rarity and in this context somewhat speculative histopathogenesis may account for diagnostic difficulties. Current classifications do not consider benign histopathological features at the primary and malignant features at the metastatic tumour site. Based on an evidence-based literature review, a recommendation for a novel classification is presented.MethodsAn evidence-based literature review over the last 60 years regarding ameloblastic carcinoma of the maxilla was conducted.ResultsAn overall of 26 cases were found (mean age: 54.4 (5-83 years); male to female ratio: 2.7 to 1). In 54% the primary diagnosis was ameloblastic carcinoma, 34.6% revealed pulmonary metastases, however, only in one patient cervical lymph node metastasis could be found. Whereas two cases did not reveal malignant histopathology at the primary, they revealed malignant features at their metastatic sites. Nineteen of 26 patients (73,1%) were controlled during a median follow-up time of 54,3 months (6 to 156 months); 6 patients died of disease after a median time of 62,7 months (7 to 156 months) after initial diagnosis.ConclusionIt is of utmost importance to be aware of that ameloblastomas may be capable to degenerate into a "malignant" disease with recurrence and metastasis. In addition to local long-term control, special attention should be paid to potential pulmonary involvement.
Highlights
Ameloblastomas, representing 1% of all jaw tumours, are considered to be benign, but locally aggressive odontogenic epithelial neoplasms [1]
44% were found in the left maxilla, and 32% in the right side of maxilla (Fig. 3); in 54% the first specimen revealed directly an ameloblastic carcinoma, but in 15% a follicular ameloblastoma was first found (Fig. 4). 26,9% revealed pulmonary metastases and in 23% local recurrence was detected (Fig. 5). 77% of the patients underwent a median follow-up period of 54.3 months (6 156 months) while 23% died of disease after a median time of 62,7 (7 months-10 years) months after initial diagnosis (Fig. 5)
In cases of maxillary ameloblastomas, a radical resection should be performed in order to prevent recurrence and development of malignancy
Summary
Ameloblastomas, representing 1% of all jaw tumours, are considered to be benign, but locally aggressive odontogenic epithelial neoplasms [1]. Amongst others, calcifying epithelial odontogenic tumor, metastatic carcinoma of the jaw and keratocystic odontogenous tumours may come into consideration as differential diagnoses. Even though ameloblastomas are well studied and documented, little is known about their malignant features. This is reflected in the fact that whereas more than 3600 cases of ameloblastomas have been described in the literature [3], fewer than 60 cases of (page number not for citation purposes). Current classifications do not consider benign histopathological features at the primary and malignant features at the metastatic tumour site. Based on an evidence-based literature review, a recommendation for a novel classification is presented
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