Diagnosis of langerhans cell histiocytosis based on maxillofacial manifestations in an adult: case report

Background: Langerhans cell histiocytosis (LCH) is a rare clonal disease that most commonly affects the pediatric population. In adults, its diagnosis is challenging due to its low prevalence and nonspecific clinical manifestations. The maxillofacial region may be the first site affected, making the dentist a key figure in early detection. Materials and Methods: We present the case of a 38-year-old male patient who consulted due to tooth mobility and difficulty chewing. Radiographic examination revealed osteolytic lesions in the mandible. The diagnosis was confirmed by biopsy with immunohistochemical positivity for CD1a, CD68, and S100. The patient was referred to the hematology-oncology team, where a systemic workup was completed. Treatment included chemotherapy with vinblastine, prednisone, and mercaptopurine, along with endocrine management and radiotherapy. Results and Discussion: The patient showed a favorable outcome, with no recurrence after four years of follow-up. The mandible was the most commonly affected site, with symptoms including pain, tooth mobility, and radiolucent lesions. Diagnosis was confirmed by immunohistochemistry in all cases. Treatment modalities varied and included surgery, radiotherapy, and chemotherapy. Discussion: This case highlights the role of dentists in identifying rare systemic diseases. Tooth mobility without evident periodontal disease may be the first sign of LCH. The literature supports a multidisciplinary approach and the need to consider LCH in the differential diagnosis of maxillofacial lesions. Conclusions: LCH can initially present in the maxillofacial region. Early recognition enables timely intervention, improving prognosis and reducing the risk of systemic complications.