{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,12]],"date-time":"2026-10-12T18:18:41Z","timestamp":1791829121328,"version":"4.3.4"},"reference-count":27,"publisher":"Wiley","issue":"2","license":[{"start":{"date-parts":[[2012,11,20]],"date-time":"2012-11-20T00:00:00Z","timestamp":1353369600000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["J Cutan Pathol"],"published-print":{"date-parts":[[2013,2]]},"abstract":"<jats:p>Langerhans cell histiocytosis (<jats:styled-content style=\"fixed-case\">LCH<\/jats:styled-content>) is characterized by a clonal proliferation of bone marrow\u2010derived Langerhans cells. While cutaneous involvement is relatively common, <jats:styled-content style=\"fixed-case\">LCH<\/jats:styled-content> restricted to the vulvar area is a rare phenomenon and can occur in different clinical settings. Occasionally, vulvar <jats:styled-content style=\"fixed-case\">LCH<\/jats:styled-content> heralds subsequent multi\u2010organ involvement with an aggressive clinical course. Even cases of <jats:styled-content style=\"fixed-case\">LCH<\/jats:styled-content> isolated to the vulvar area can present with local recurrences despite excision and radiation. We present a case of a 68\u2010year\u2010old female with a 1\u2010month history of pruritic lesions on her vulva. Physical examination showed whitish plaques with scattered nodular areas on the labia majora. A vulvar biopsy showed a background of lichen sclerosus  (LS) with foci of oval to polygonal cells with moderately abundant eosinophilic cytoplasm and folded nuclei showing frequent nuclear grooves. Immunohistochemical staining showed that the cells were positive for <jats:styled-content style=\"fixed-case\">CD1a<\/jats:styled-content> and S\u2010100, confirming the diagnosis of <jats:styled-content style=\"fixed-case\">LCH<\/jats:styled-content>. On further workup, there was no evidence of disseminated disease involving other organs. While vulvar <jats:styled-content style=\"fixed-case\">LCH<\/jats:styled-content> is uncommonly seen, and with only one previous case report in the literature associated in the setting of lichen sclerosus, this case illustrates the importance of recognizing this condition and ensuring proper clinical follow\u2010up to rule out a systemic involvement.<\/jats:p>","DOI":"10.1111\/cup.12051","type":"journal-article","created":{"date-parts":[[2012,11,20]],"date-time":"2012-11-20T14:36:26Z","timestamp":1353422186000},"page":"279-283","source":"Crossref","is-referenced-by-count":10,"title":["Langerhans cell histiocytosis associated with lichen sclerosus of the vulva: case report and review of the literature"],"prefix":"10.1111","volume":"40","author":[{"given":"Jason C.","family":"Chang","sequence":"first","affiliation":[{"name":"Department of Pathology Medical College of Wisconsin  Milwaukee WI USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"David 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