Systematic Reviews
Copyright ©The Author(s) 2021. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Oncol. Dec 24, 2021; 12(12): 1227-1243
Published online Dec 24, 2021. doi: 10.5306/wjco.v12.i12.1227
Mixed odontogenic tumors: A review of the clinicopathological and molecular features and changes in the WHO classification
Celeste Sánchez-Romero, Oslei Paes de Almeida, Ronell Bologna-Molina
Celeste Sánchez-Romero, Ronell Bologna-Molina, Department of Oral Pathology, Faculty of Dentistry, University Juarez of the Durango State, Durango 33106, Mexico
Oslei Paes de Almeida, Department of Diagnosis, Faculty of Piracicaba, State University of Campinas, São Paulo Brazil, Piracicaba 13414-903, Sao Paulo, Brazil
Ronell Bologna-Molina, Department of Oral and Maxillofacial Pathology, Universidad de la República UDELAR (URUGUAY), Montevideo 16400, Uruguay
Author contributions: Sánchez-Romero C, Bologna-Molina R and Paes de Almeida O participated in the conceptualization, bibliographic search, selection of information, interpretation of data, writing of the manuscript as well as in the subsequent revision of the manuscript.
Conflict-of-interest statement: All the authors have indicated that they have no potential conflicts of interest and no financial relationships relevant to this article to disclose.
PRISMA 2009 Checklist statement: The authors have read the PRISMA 2009 Checklist, and the manuscript was prepared and revised according to the PRISMA 2009 Checklist.
Open-Access: This article is an open-access article that was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution NonCommercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See: https://creativecommons.org/Licenses/by-nc/4.0/
Corresponding author: Ronell Bologna-Molina, DDS, MSc, PhD, Professor, Department of Oral and Maxillofacial Pathology, Universidad de la República UDELAR (URUGUAY), Montevideo 16400, Uruguay. ronellbologna@hotmail.com
Received: January 19, 2021
Peer-review started: January 19, 2021
First decision: May 14, 2021
Revised: May 25, 2021
Accepted: November 25, 2021
Article in press: November 25, 2021
Published online: December 24, 2021
Processing time: 339 Days and 6.9 Hours
Abstract
BACKGROUND

Ameloblastic fibromas and ameloblastic fibrosarcomas are rare odontogenic tumors, and controversy exists in the classification of cases presenting hard-tissue production: Ameloblastic fibrodentinoma (AFD) and ameloblastic fibro-odontoma (AFO). These cases are currently considered “developing odontomas” (hamartomatous lesions).

AIM

To analyze the clinicopathologic features of these lesions and discuss the changes in the 2017 World Health Organization classification.

METHODS

An electronic literature search was performed in the PubMed/MEDLINE database. An electronic search of the English language literature was performed and last updated in September 2020 in the PubMed/MEDLINE database using the following terms: “ameloblastic fibroma”, “ameloblastic fibrodentinoma”, “ameloblastic fibro-odontoma”, “ameloblastic sarcoma”, “ameloblastic fibrosarcoma”, “ameloblastic fibrodentinosarcoma”, “ameloblastic fibroodontosarcoma” and “odontogenic carcinosarcoma”. The inclusion criteria were odontogenic tumor series, case reports and systematic reviews that provided sufficient clinical, radiological and microscopic documentation to confirm the diagnosis.

RESULTS

The database search strategy resulted in 947 papers. Articles focusing on other topics, articles that were not in English, duplicate articles, and articles without fulfilling the inclusion criteria were excluded. Finally, 96 publications were included in this review to describe and discuss the main features of the searched entities. Several aspects of AFO and AFD, such as biological behavior, age of occurrence, amount of hard tissue, and potential for malignant transformation into odontogenic sarcomas, support the neoplastic nature in most of the reported cases. Considering the clinical, radiographic, histopathological and molecular characteristics of odontogenic lesions with hard tissue production, we suggest that these types of lesions should continue to be recognized as odontogenic tumors by maintaining the classically used terms.

CONCLUSION

This recommendation will be relevant for future clinical, microscopic, and molecular studies to better understand the biology of these interesting odontogenic tumors.

Keywords: Ameloblastic fibroma; Ameloblastic fibrosarcoma; Odontogenic carcinosarcoma; Odontogenic tumors

Core Tip: We consider that the recent 2017 World Health Organization classification does not clarify the subject when considering ameloblastic fibrodentinoma (AFD) and ameloblastic fibro-odontoma (AFO) as “developing odontomas”. According to the clinical, radiographical, histopathological and molecular features of the cases reviewed, we suggest that AFD and AFO should continue to be considered benign neoplasms. Thus, the nomenclature of these mixed benign odontogenic tumors would be congruent with the classification of ameloblastic/odontogenic sarcomas. Additionally, further studies are warranted to compare these interesting odontogenic tumors and finally better clarify and understand their similarities and differences.