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Case Reports in Pathology
Volume 2017, Article ID 1691403, 6 pages
Case Report

Langerhans Cell Histiocytosis: A Diagnostic Challenge in the Oral Cavity

1Department of Oral and Maxillofacial Surgery, Akdeniz University, Faculty of Dentistry, Antalya, Turkey
2Department of Pathology, Akdeniz University, Faculty of Medicine, Antalya, Turkey
3Department of Hematology, Hatay State Hospital, Hatay, Turkey
4Department of Hematology, Akdeniz University, Faculty of Medicine, Antalya, Turkey
5Department of Oral and Maxillofacial Surgery, Case Western Reserve University, School of Dental Medicine, Cleveland, OH, USA

Correspondence should be addressed to Mehmet Ali Altay; moc.liamtoh@yatlailam

Received 30 June 2017; Accepted 3 October 2017; Published 24 October 2017

Academic Editor: Achille Pich

Copyright © 2017 Mehmet Ali Altay 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.


Background. Langerhans cell histiocytosis (LCH) is a rare disorder of the reticuloendothelial system with unknown etiology. This report aims to present a case of LCH with diffuse involvement of the oral cavity and to raise awareness of the distinguishing features of this diagnostically challenging entity. Case Report. A 26-year-old male patient presented with complaints of teeth mobility, intense pain, and difficulty in chewing. Intraoral and radiological examinations revealed generalized gingival hyperplasia and severe teeth mobility with widespread alveolar bone loss. Periodontal therapy was performed with no significant improvement. An incisional biopsy revealed Langerhans cells and positive reaction to S-100 and CD1, and the patient was diagnosed with LCH. The patient underwent systemic chemotherapy with vinca alkaloids and corticosteroids. Regression of gingival lesions, as well as significant decrease in mobility of the remaining teeth and severity of pain, was achieved during 12 months of follow-up. Conclusion. The rarity and variable system involvement of LCH necessitate a multidisciplinary approach be carried out for accurate diagnosis, effective treatment, and an uneventful follow-up. Awareness of oral manifestations of LCH may aid clinicians greatly in reducing morbidity and mortality associated with this debilitating condition.