Case Report Lingual Leiomyomatous Hamartoma in an Adult Male Amanda Phoon Nguyen , 1 Norman Firth, 1 Sophie Mougos, 2 and Omar Kujan 1 1 UWA Dental School, University of Western Australia, Nedlands, WA 6009, Australia 2 Private Practice, OMFSurgery, Cambridge Street, Wembley, WA, Australia Correspondence should be addressed to Amanda Phoon Nguyen; phoonamanda@gmail.com Received 6 March 2018; Accepted 29 July 2018; Published 29 August 2018 Academic Editor: Konstantinos Michalakis Copyright © 2018 Amanda Phoon Nguyen et al. This is an open access article distributed under the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited. An otherwise healthy 20-year-old male presented with an exophytic, polypoid, yellowish lesion involving the dorsal surface of his tongue, which he reported being present since birth and unchanged. This was removed by surgical excision and diagnosed as a leiomyomatous hamartoma. Histological examination revealed a combination of fibrovascular connective tissue, conspicuous smooth-muscle bundles, adipose tissue, minor salivary gland tissue, blood vessels, lymphoid tissue, peripheral nerves, and normal skeletal muscle. This case is exceptional due to the patient’s age, as until now, lingual leiomyomatous hamartomas have been reported almost exclusively in a paediatric population. To our knowledge, this is the eldest age at which a LLH has been reported in the literature. This underscores the need for clinicians to consider this rarely reported entity when considering the radiographic and clinical differential diagnoses for these lesions, both in the paediatric and adult populations. We also present a review of the literature regarding lingual leiomyomatous hamartomas. 1. Introduction The term hamartoma is used to describe a nonneoplastic, abnormal, and haphazard overgrowth of conglomerates of mature cells and tissues indigenous to the anatomic site from which it occurs, often with one predominating element [1, 2]. This tumor-like malformation is benign, and most hamarto- mas are located in the liver, spleen, pancreas, and kidneys [3]. Oral hamartomas are rare [3]. An oral leiomyomatous hamartoma, as the name suggests, is therefore a lesion com- posed mostly of smooth-muscle tissue. They typically present as smooth, soft, nodular lesions that are present at birth and further develop in the first decade of life [3]. They are usually painless masses without obvious symptoms [1]. In most of the published reports, manifestations have commonly been on the gingiva, tongue, and hard palate, specifically the dorsum of the tongue and the anterior maxillary gingiva or alveolar ridge in the incisive papilla region [1, 3]. Due to its low clinical morbidity and nonspecific symptoms, diagnosis and treatment remains a challenge [4]. Here, we report an unusual case of a lingual leiomyoma- tous hamartoma (LLH) occurring in a healthy 20-year-old male, treated successfully by surgical excision. We also pres- ent the histological and immunohistochemical features as well as a review of the literature. This case is exceptional due to the patient’s age, as until now, LLHs have been reported almost exclusively in a paediatric population. To our knowledge, this is the eldest age at which a LLH has been reported in the literature. 2. Case Report A 20-year-old medically fit and healthy male presented for an assessment in preparation for orthognathic surgery. On examination, a 1.5 cm diameter exophytic midline tongue lesion (Figure 1) was noted. This lesion was smooth, regular, and soft to palpation. He reported that this had been present since birth with no change since childhood. A magnetic res- onance image (MRI) of his tongue was obtained (Figure 2). Hindawi Case Reports in Dentistry Volume 2018, Article ID 4162436, 5 pages https://doi.org/10.1155/2018/4162436