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Successful treatment of adult's Langerhans cell histiocytosis with thalidomide. Report of two cases and literature review.

REPORT OF A CASE CASE 1 A 59-year-old woman treated for diabetes insipidus by desmopressin (Minirin) for 8 years was seen with large granulomatous ulcerations and infiltrated papules of the perianal region that were present for 3 years ( Figure 1 ). A few papular lesions were seen in the submammary region. Histopathologic examination of a skin biopsy specimen taken from the perianal lesions disclosed a thick dermal infiltrate with lymphocytes, eosinophils, and large histiocytes ( Figure 2 ). Immunohistopathologic study showed that the histiocytes stained positive for S100 and CD la. Electron microscopy confirmed the diagnosis of Langerhans cell histiocytosis, showing 20% of the cells in the infiltrate contained Birbeck's granules ( Figure 3 ). The patient's clinical examination showed negative findings, and no pulmonary involvement was seen on a chest roentgenogram and a thoracic computed tomographic scan. The bone marrow biopsy specimen showed normal findings. Endocrine tests revealed deficiences in corticotropin, gondatropin, and somatotropin