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We report the case of a 2\u2010year\u2010old 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\u2010infected B\u2010cells present in lesional tissue implying that interactions between EBV\u2010infected B\u2010cells and lesional Langerhans cells may be associated with the development of LCH. Pediatr Blood Cancer 2008;50:924\u2013927. \u00a9 2007 Wiley\u2010Liss, Inc.<\/jats:p>","DOI":"10.1002\/pbc.21249","type":"journal-article","created":{"date-parts":[[2007,5,1]],"date-time":"2007-05-01T19:55:29Z","timestamp":1178049329000},"page":"924-927","source":"Crossref","is-referenced-by-count":22,"title":["Development of Langerhans cell histiocytosis associated with chronic active Epstein\u2013Barr virus infection"],"prefix":"10.1002","volume":"50","author":[{"given":"Naoki","family":"Sakata","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Naomi","family":"Toguchi","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Masatomo","family":"Kimura","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Masahiro","family":"Nakayama","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Keisei","family":"Kawa","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Tsukasa","family":"Takemura","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"311","published-online":{"date-parts":[[2007,5]]},"reference":[{"key":"e_1_2_5_2_2","doi-asserted-by":"publisher","DOI":"10.1056\/NEJM200008173430707"},{"key":"e_1_2_5_3_2","doi-asserted-by":"publisher","DOI":"10.1016\/0167-5699(86)90183-0"},{"key":"e_1_2_5_4_2","doi-asserted-by":"crossref","first-page":"132","DOI":"10.1128\/JCM.37.1.132-136.1999","article-title":"Quantitative analysis of Epstein\u2013Barr virus load by using a real\u2010time PCR assay","volume":"37","author":"Kimura H","year":"1999","journal-title":"J Clin Microbiol"},{"key":"e_1_2_5_5_2","doi-asserted-by":"publisher","DOI":"10.1056\/NEJM198803243181203"},{"key":"e_1_2_5_6_2","doi-asserted-by":"publisher","DOI":"10.1038\/333455a0"},{"key":"e_1_2_5_7_2","doi-asserted-by":"crossref","first-page":"2085","DOI":"10.1182\/blood.V91.6.2085","article-title":"A syndrome of peripheral blood T\u2010cell infection with Epstein\u2013Barr virus (EBV) followed by EBV\u2010positive T\u2010cell lymphoma","volume":"91","author":"Kanegane H","year":"1998","journal-title":"Blood"},{"key":"e_1_2_5_8_2","first-page":"3","article-title":"Langerhans' cell histiocytosis pathobiology and pathogenesis","volume":"18","author":"Favara BE","year":"1991","journal-title":"Semin Oncol"},{"key":"e_1_2_5_9_2","doi-asserted-by":"publisher","DOI":"10.1172\/JCI200421140"},{"key":"e_1_2_5_10_2","doi-asserted-by":"publisher","DOI":"10.1056\/NEJM199407213310303"},{"key":"e_1_2_5_11_2","doi-asserted-by":"publisher","DOI":"10.1016\/S0140-6736(94)91842-2"},{"key":"e_1_2_5_12_2","doi-asserted-by":"publisher","DOI":"10.1007\/s00431-004-1493-y"},{"key":"e_1_2_5_13_2","doi-asserted-by":"publisher","DOI":"10.1002\/jor.20039"},{"key":"e_1_2_5_14_2","doi-asserted-by":"publisher","DOI":"10.1002\/ajh.10004"},{"key":"e_1_2_5_15_2","doi-asserted-by":"publisher","DOI":"10.1111\/j.1349-7006.1990.tb02561.x"},{"key":"e_1_2_5_16_2","doi-asserted-by":"publisher","DOI":"10.1007\/BF02983236"},{"key":"e_1_2_5_17_2","doi-asserted-by":"publisher","DOI":"10.1532\/IJH97.05083"},{"key":"e_1_2_5_18_2","doi-asserted-by":"publisher","DOI":"10.1002\/ajh.2830470104"},{"key":"e_1_2_5_19_2","doi-asserted-by":"publisher","DOI":"10.1016\/j.humpath.2004.03.017"},{"key":"e_1_2_5_20_2","doi-asserted-by":"publisher","DOI":"10.1016\/S0959-8049(00)00296-3"},{"key":"e_1_2_5_21_2","doi-asserted-by":"publisher","DOI":"10.1073\/pnas.1231363100"}],"container-title":["Pediatric Blood &amp; Cancer"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.wiley.com\/onlinelibrary\/tdm\/v1\/articles\/10.1002%2Fpbc.21249","content-type":"unspecified","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/pbc.21249","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,10,12]],"date-time":"2023-10-12T03:52:33Z","timestamp":1697082753000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1002\/pbc.21249"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2007,5]]},"references-count":20,"journal-issue":{"issue":"4","published-print":{"date-parts":[[2008,4]]}},"alternative-id":["10.1002\/pbc.21249"],"URL":"https:\/\/doi.org\/10.1002\/pbc.21249","archive":["Portico"],"relation":{},"ISSN":["1545-5009","1545-5017"],"issn-type":[{"value":"1545-5009","type":"print"},{"value":"1545-5017","type":"electronic"}],"subject":[],"published":{"date-parts":[[2007,5]]}}}