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October 1993

Successful Treatment of Adult's Langerhans Cell Histiocytosis With Thalidomide: Report of Two Cases and Literature Review

Author Affiliations

Hôpital de l'Antiquaille, Lyon, France

Arch Dermatol. 1993;129(10):1261-1264. doi:10.1001/archderm.1993.01680310031004


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

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