Calcifying epithelial odontogenic tumor: An updated analysis of 339 cases reported in the literature Bruno Ramos Chrcanovic a, * , Ricardo Santiago Gomez b a Department of Prosthodontics (Head: Dr. Ann Wennerberg, DDS, PhD), Faculty of Odontology, Malm€ o University, Malm€ o, Sweden b Department of Oral Surgery and Pathology (Head: Dr. Henrique Pretti), School of Dentistry, Universidade Federal de Minas Gerais, Belo Horizonte, Brazil article info Article history: Paper received 6 February 2017 Accepted 3 May 2017 Available online 12 May 2017 Keywords: Calcifying epithelial odontogenic tumor Pindborg tumor Intraosseous Extraosseous Clinical features Recurrence rate abstract Purpose: The aim of this study was to integrate the available data published on calcifying epithelial odontogenic tumors (CEOT) into a comprehensive analysis of its clinical and radiologic features. Materials and methods: An electronic search was undertaken in May 2016. Eligibility criteria were pub- lications having enough clinical, radiological and/or histological information to confirm definite diagnosis. Results: A total of 362 lesions were found, 339 with enough information were analyzed. Variants clear cells (n ¼ 33) and Langerhans cells (n ¼ 10) were rarely described in the literature, as well as lesions with malignant transformation (n ¼ 8). Central lesions (n ¼ 264) were more prevalent than their peripheral counterparts (n ¼ 24). A higher prevalence characterized the mandible, posterior region, and third and fourth decades. About 40% of the peripheral lesions showed signs of underlying bone erosion, and about half of the central ones showed signs of cortical bone perforation. Recurrence was found in all lesions (12.6%), peripheral lesions (18.8%), central lesions (11.6%), clear cell (10.7%), Langerhans cell (0%), and those with malignant transformation (42.9%). Excision or curettage was associated with the highest recurrence rate. None of the variables showed a statistically significant influence on the recurrence rate. Conclusions: The possible locally aggressive behavior of the lesions recommends a less conservative management than simple curettage. The clear cell variant shows similar demographic data and biological behavior compared to the non-variant lesions, suggesting that the presence of clear cells does not have an important clinical significance. © 2017 European Association for Cranio-Maxillo-Facial Surgery. Published by Elsevier Ltd. All rights reserved. 1. Introduction The calcifying epithelial odontogenic tumor (CEOT), also called the Pindborg tumor, is an epithelial odontogenic neoplasm first reported by Thoma and Goldman (1946) some 70 years ago. CEOT is predominantly an intraosseous (central) neoplasm, but it also oc- curs as a rare, less aggressive peripheral (extraosseous) tumor (Afrogheh et al., 2014). Two variants of CEOT have been described in the literature: the clear cell CEOT and the Langerhans cell CEOT, first reported in 1967 by Abrams and Howell (1967) and in 1990 by Asano et al. (1990), respectively. Cases of CEOT are not so often reported in the literature. Epidemiological study of such rare lesions is of great importance because it provides information that can improve the diagnostic accuracy and will allow pathologists and surgeons to make informed decisions and refine the treatment plan to optimize the clinical outcome. The aim of the present study was to integrate the available data published in the literature on CEOT into an updated comprehensive analysis of its clinical and radiologic features, as well as the frequency of recurrence. 2. Materials and methods This study followed the PRISMA Statement guidelines (Moher et al., 2009). A review protocol does not exist. 2.1. Search strategies An electronic search without time restrictions was undertaken in May 2016 in the following databases: PubMed/Medline, Web of Science, and Science Direct. The following terms were used in the * Corresponding author. Department of Prosthodontics, Faculty of Odontology, Malm€ o University, Carl Gustafs v€ ag 34, SE-214 21, Malm€ o, Sweden. Fax: þ46 40 6658503. E-mail addresses: brunochrcanovic@hotmail.com, bruno.chrcanovic@mah.se (B.R. Chrcanovic), rsgomez@ufmg.br (R.S. Gomez). Contents lists available at ScienceDirect Journal of Cranio-Maxillo-Facial Surgery journal homepage: www.jcmfs.com http://dx.doi.org/10.1016/j.jcms.2017.05.007 1010-5182/© 2017 European Association for Cranio-Maxillo-Facial Surgery. Published by Elsevier Ltd. All rights reserved. Journal of Cranio-Maxillo-Facial Surgery 45 (2017) 1117e1123