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Development of Langerhans cell histiocytosis associated with chronic active Epstein–Barr virus infection

Chronic active Epstein–Barr virus (CAEBV) infection is characterized by a status of lymphoproliferative disease of EBV‐infected cells, resulting in chronic or recurrent infectious mononucleosis‐like symptoms. CAEBV is always accompanied by life‐threatening complications. We report the case of a 2‐year‐old female patient with CAEBV who subsequently developed Langerhans cell histiocytosis (LCH) presenting with bilateral exophthalmos, bone, and skin involvement. In situ hybridization for EBER revealed EBV‐infected B‐cells present in lesional tissue implying that interactions between EBV‐infected B‐cells and lesional Langerhans cells may be associated with the development of LCH. Pediatr Blood Cancer 2008;50:924–927. © 2007 Wiley‐Liss, Inc.