A 19-year-old Thai man presented with symptoms of central diabetes insipidus, exopthalmos and multiple ostolytic lesions of the cranial vault. Skin biopsy showed strongly positive CD 1a and S100. Electron microscopy showed rod shape and pentalaminar granules called "Birbeck" granules, a diagnosis compatible with Langerhans' cell histiocytosis (LCHs) or histiocytosis X. He was successfully treated with systemic chemotherapy.
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