Mandibular Langerhans Cell Histiocytosis in Pediatric and Geriatric Patients: Report of Two Cases with Contrasting Clinical Features

Langerhans Cell Histiocytosis (LCH) is a rare clonal proliferative disorder of dendritic cells that most commonly affects children but may also present in adults and elderly patients. When the mandible is involved, its clinical and radiographic appearance often mimics a variety of benign or malignant jaw lesions, posing significant diagnostic challenges for dental and medical practitioners. In this report, we describe two illustrative cases of mandibular LCH occurring at opposite ends of the age spectrum. The first case involved a 3-year-old girl who presented with a painless swelling in the anterior mandible. Radiographic examination revealed a poorly defined radiolucent lesion, and histopathological analysis with molecular testing confirmed LCH harboring the BRAF V600E mutation. The patient received targeted therapy with Vemurafenib, resulting in substantial clinical improvement and sustained remission during follow-up. The second case concerned a 65-year-old male who presented with persistent mandibular pain, lower-lip paresthesia, and an intraoral ulcer. Imaging demonstrated multiple bilateral punched-out radiolucencies in the mandible. Histopathological and immunohistochemical studies confirmed the diagnosis of LCH. The patient underwent surgical segmental resection of the affected mandible with subsequent reconstruction using a titanium plate, leading to satisfactory healing and no evidence of recurrence on follow-up. These contrasting cases emphasize the importance of considering LCH in the differential diagnosis of mandibular lesions across all age groups. Prompt imaging, biopsy, molecular testing, and multidisciplinary management are crucial for accurate diagnosis and effective treatment strategies that improve patient outcomes.

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Publication Details

Journal
International Journal of Clinical Oral and Maxillofacial Surgery
Published
2025-10-28
DOI
https://doi.org/10.11648/j.ijcoms.20251102.12
Primary Topic
Histiocytic Disorders and Treatments
Type
article
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article

Mandibular Langerhans Cell Histiocytosis in Pediatric and Geriatric Patients: Report of Two Cases with Contrasting Clinical Features

International Journal of Clinical Oral and Maxillofacial Surgery
Histiocytic Disorders and Treatments
article

Mandibular Langerhans Cell Histiocytosis in Pediatric and Geriatric Patients: Report of Two Cases with Contrasting Clinical Features

article en

Abstract

Langerhans Cell Histiocytosis (LCH) is a rare clonal proliferative disorder of dendritic cells that most commonly affects children but may also present in adults and elderly patients. When the mandible is involved, its clinical and radiographic appearance often mimics a variety of benign or malignant jaw lesions, posing significant diagnostic challenges for dental and medical practitioners. In this report, we describe two illustrative cases of mandibular LCH occurring at opposite ends of the age spectrum. The first case involved a 3-year-old girl who presented with a painless swelling in the anterior mandible. Radiographic examination revealed a poorly defined radiolucent lesion, and histopathological analysis with molecular testing confirmed LCH harboring the BRAF V600E mutation. The patient received targeted therapy with Vemurafenib, resulting in substantial clinical improvement and sustained remission during follow-up. The second case concerned a 65-year-old male who presented with persistent mandibular pain, lower-lip paresthesia, and an intraoral ulcer. Imaging demonstrated multiple bilateral punched-out radiolucencies in the mandible. Histopathological and immunohistochemical studies confirmed the diagnosis of LCH. The patient underwent surgical segmental resection of the affected mandible with subsequent reconstruction using a titanium plate, leading to satisfactory healing and no evidence of recurrence on follow-up. These contrasting cases emphasize the importance of considering LCH in the differential diagnosis of mandibular lesions across all age groups. Prompt imaging, biopsy, molecular testing, and multidisciplinary management are crucial for accurate diagnosis and effective treatment strategies that improve patient outcomes.

International Journal of Clinical Oral and Maxillofacial SurgeryVol. 11(2)
Urmia University (IR), Urmia University of Medical Sciences (IR)
Good health and well-being
Openalex Percentile: Top 39%
Histiocytic Disorders and Treatments
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Mandibular Langerhans Cell Histiocytosis in Pediatric and Geriatric Patients: Report of Two Cases with Contrasting Clinical Features · International Journal of Clinical Oral and Maxillofacial Surgery (2025) | TGRS Research Map | TGRS