Abstract
Background : Langerhans cell histiocytosis (LCH) is a lesion characterized by proliferation of Langerhans cells and comparatively rarely affects the mandible. We report herein on a case of LCH in which preoperative cytological diagnosis suggested LCH.
Case : A two-year-old boy was admitted with swelling of the right cheek. Computed tomography demonstrated a broad bone expansion and defect extending from the right mandibular molar to the ramus region. Scintigraphy revealed no increased uptake except in the mandibular region. The preoperative aspiration cytology specimen revealed eosinophils and large histiocyte-like cells having indented or lobulated nuclei. The surgical specimen showed proliferation of Langerhans cells with eosinophilic infiltration. On immunohistochemical staining, the Langerhans cells were positive for S-100 protein, vimentin, CD1a, langerin and HLA-DR. Electron microscopy showing Birbeck granules in the cytoplasm of Langerhans cells thus revealed the diagnosis of LCH.
Conclusion : Although we did not confirm a definitive diagnosis cytologically in this case, future accumulation of data from cases of LCH with preoperative cytology will lead to an accurate diagnosis.
