Malignant Odontogenic Tumours: A Systematic Review of Cases Reported in Literature

Abstract

Background: Malignant odontogenic tumours (MOTs) arise either de novo from the tooth forming tissues, their developmental residues or from existing odontogenic epithelial or mesenchymal neoplasms in the jaws. Their management requires extensive surgery due to their infiltrative nature and risk of metastasis. There is a need to understand the clinical and pathological features of MOTs to inform both treatment algorithms and prognostication. This is an area of diagnostic pathology which presents substantial difficulties in diagnosis, compounded by inconsistent use of terminology. Thus, this systematic review aimed to describe the clinical and pathological features of MOTs with a view to consolidating the literature and defining problematic areas in diagnosis and classification.Methods: An electronic database search was conducted in Web of Science, PubMed/Medline, and Embase. Additionally, the grey literature and reference lists of selected papers searched for completeness. Nine hundred and sixty articles were initially identified. Following removal of duplicates and application of inclusion/exclusion criteria, 312 articles were included for qualitative analysis.Results: The 312 articles encompassed a total of 507 patients with most lesions located within the mandible (74.3%). The most common first histological diagnosis was ameloblastic carcinoma (25.7% of all diagnoses), but there is considerable variation in how and when various diagnostic terms are used, and several misdiagnoses were reported. An initial benign diagnosis was made in 24.7% of patients, followed by a later malignant diagnosis and in this sub-group, the most common benign first diagnosis was ameloblastoma (42.4%). Cervical lymph nodes were the most common site of metastasis (9.3% of patients). With respect to distant metastasis (DM), the lungs were the most common organ affected (11.2% of DM patients) with metastasising ameloblastoma the most commonly reported tumour which metastasised to the lungs. Overall, 26.8% of patients developed recurrence.Conclusion: Overall, the quality of the literature on MOTs is poor. This review of the literature has highlighted variations in diagnostic terms and criteria which has resulted in areas of confusion with potential for misdiagnosis. This consolidation of primary data has identified key areas for targeted research including further discussion on the malignant potential of ameloblastoma.

BRAF V600E Mutation in Ameloblastoma: A Systematic Review and Meta-Analysis

“…There was also a case report which was ameloblastoma initially but later presented with ameloblastic carcinoma; positive BRAF V600E mutation was subsequently detected [ 99 ]. This malignant counterpart also expresses BRAF V600E mutation (about 25 to 33%) [ 100 , 101 ]. Hence, there is a possibility that BRAF V600E mutation may play a role in the malignant transformation of ameloblastoma, as this rare odontogenic malignancy has close features that combine the histologic features of ameloblastoma with cytologic atypia [ 100 , 101 , 102 ].…”

Desmoplastic Adamantinoma-like Thymic Carcinoma

“…The differential diagnosis for these lesions is quite limited. Metastases from ameloblastoma are extremely rare and most frequently involve the lungs 60. Metastases are more common in cases showing overt evidence of malignant transformation (ie, ameloblastic carcinoma).…”

“…Metastases from ameloblastoma are extremely rare and most frequently involve the lungs. 60 Metastases are more common in cases showing overt evidence of malignant transformation (ie, ameloblastic carcinoma). Ameloblastic carcinomas are quite rare and represent <1% of all odontogenic tumors, and are known to develop pulmonary and cervical lymph node metastases in about a third of cases; they can appear histologically indistinguishable from ameloblastoma other than for increased mitotic activity.…”

A Current Review of Machine Learning and Deep Learning Models in Oral Cancer Diagnosis: Recent Technologies, Open Challenges, and Future Research Directions

“…Odontogenic carcinosarcoma (OCS) is a very rare form of jaw cancer that is made up of both epithelial and mesenchymal components [ 87 , 88 ]. It is similar in structure to ameloblastic fibroma (AF) and can appear as a new lesion or develop from an existing odontogenic lesion [ 87 , 89 ]. The histological features of OCS include malignant epithelial cells present in hypercellular mesenchymal tissue, and hyperchromatic and pleomorphic cells.…”

“…The histological features of OCS include malignant epithelial cells present in hypercellular mesenchymal tissue, and hyperchromatic and pleomorphic cells. OCS can be mistaken for other types of odontogenic tumors and has a high rate of recurrence and metastases [ 89 ]. The WHO initially classified OCS as a unique entity in 1992, but later withdrew it in the third edition of its classification in 2005 due to difficulty in distinguishing it from other tumors; it was later reinstated as a unique entity in the most recent classification by WHO in 2017 [ 90 , 91 ].…”