International Journal of Research in Medical Sciences | April 2023 | Vol 11 | Issue 4 Page 1383 International Journal of Research in Medical Sciences Vichattu SV et al. Int J Res Med Sci. 2023 Apr;11(4):1383-1387 www.msjonline.org pISSN 2320-6071 | eISSN 2320-6012 Case Report Extrafollicular adenomatoid odontogenic tumor: a case report Sankar Vinod Vichattu, Arun George, Joy R. Das, Atic Thomas, Athira K. Sasi* INTRODUCTION AOT is not a new entity in the literature, World Health Organization (WHO) in 1971 adopted the term ‘adenomatoid odontogenic tumor’ till then various terminologies were used to describe AOT. The name adenomatoid is given because histologically the tumor shows numerous duct-like structures interposed within the lesion giving rise to a glandular or adenomatoid appearance. Terminologies to describe AOT evolved from epithelioma adamantium by Steenslandin in 1905, pseudo-adamantium by Dreybladt in 1907, glandular adamantinoma by L'Esperance in 1910, and cystic adamantoma by Harbitz in 1915. Stafne in 1948 coined it as a distinct entity, still others described it as an ameloblastoma variant. Philipsen and Birn suggested ‘adenomatoid odontogenic tumor’ terminology and WHO later adopted it. 1 Odontogenic tumors were classified primarily based on whether they are biologically benign or malignant. 2005 classification divided the benign tumors into “Odontogenic epithelium with odontogenic ectomesenchyme, with or without hard tissue formation, Odontogenic epithelium without odontogenic ectomesenchyme, and Odontogenic ectomesenchyme with or without odontogenic epithelium.” AOT is categorized under odontogenic epithelium with odontogenic ectomesenchyme, with or without hard tissue formation subclass. While accurate, this seemed overly complex and the 2017 version recognized epithelial, mesenchymal, and mixed tumors. Now it is categorized under benign tumor of epithelial origin. 2 CASE REPORT A 22-year female patient reported a chief complaint of swelling in the upper right front tooth region since one month. Swelling increased in size within a short duration. No pain, pus discharge, and trauma history were reported. Extra oral examination unveiled a diffuse solitary swelling of size 3.5×4 cm, with a shiny surface extending anteroposteriorly from the lateral aspect of the nose to about 4 cm in front of tragus and superoinferiorly approximately 1 cm below the infraorbital rim to right corner of mouth, also 1cm above with nasolabial fold obliteration and lifting right ala of nose causing gross facial asymmetry. On palpation, firm consistency, Department of Oral and Maxillofacial Surgery, Mar Baselios Dental College, Kothamangalam, Kerala, India Received: 16 November 2022 Revised: 08 March 2023 Accepted: 09 March 2023 *Correspondence: Dr. Athira K. Sasi, E-mail: athiraks1996@gmail.com Copyright: © the author(s), publisher and licensee Medip Academy. This is an open-access article distributed under the terms of the Creative Commons Attribution Non-Commercial License, which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited. ABSTRACT Adenomatoid odontogenic tumor (AOT), a benign epithelial odontogenic tumor, constitutes around 2-7% of all odontogenic tumors that are biopsied. This lesion is unique with regard to its clinical, radiographic, and histopathological features making its diagnosis more predictable. This article emphasizes an extrafollicular variant of AOT leading to maxillary anterior jaw swelling along with cortical plate expansion, perforation, and root resorption within a short duration of time pointing towards its aggressive nature. Keywords: Adenomatoid odontogenic tumor, Extrafollicular variant, Enucleation, Root resorption DOI: https://dx.doi.org/10.18203/2320-6012.ijrms20230897