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VOLUME 12 , ISSUE 2 ( May-August, 2024 ) > List of Articles

CASE REPORT

Langerhans Cell Histiocytosis of the Oral Cavity: A Rare Case Report and Diagnostic Dilemma

Sarika Kapila, Tejinder Kaur, Advaya Rath, Utkarsha Kapila

Keywords : Case report, Langerhans cell histiocytosis, Mobile teeth, Rare disease, Systemic involvement

Citation Information : Kapila S, Kaur T, Rath A, Kapila U. Langerhans Cell Histiocytosis of the Oral Cavity: A Rare Case Report and Diagnostic Dilemma. 2024; 12 (2):121-124.

DOI: 10.5005/djas-11014-0052

License: CC BY-NC-ND 4.0

Published Online: 31-08-2024

Copyright Statement:  Copyright © 2024; The Author(s).


Abstract

Langerhans cell histiocytosis (LCH) is a peculiar derangement regulated by cells of the reticuloendothelial system that may sometimes involve the oral cavity. The purpose of the present report is to suggest a case of LCH manifesting in the cavities of the mouth and how it may present as a diagnostic challenge. A female patient aged 33 reported having complaints of loose teeth, bleeding gums and foul smell from the mouth. Clinical and radiographic examination revealed multiple areas of gingival necrosis, ulcerations and aggressive bone loss. Initially diagnosed as aggressive periodontitis, when the lesions did not resolve, an incisional biopsy along with immunohistochemistry found Langerhans cells, affirmative reciprocation to langerin as well as CD1a, subsequently the female patient was clinically diagnosed as positive for LCH. The patient was given chemotherapy supplemented with Vinca alkaloids coupled with corticosteroids through a systemic route. The patient had multiorgan involvement, but successive retrogradation of lesions lying in the gingival area, along with a marked reduction in overall tooth mobility and harshness of pain perception, observed on the 6-month follow-up. Conclusion: Langerhans cell histiocytosis is a uniquely rare malady that may sometimes manifest in the oral cavity, so precise detection using appropriate clinical diagnostic methods, treatment and none-relapse follow-up are essential for adequate management. Awareness regarding oral manifestations of LCH is indispensable in clinical practice, as it not only aids in timely diagnosis but also encourages early intervention, thereby providing a better prognosis for the affected subject cases.


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