{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,18]],"date-time":"2026-08-18T14:18:07Z","timestamp":1787062687675,"version":"build-2736575974"},"reference-count":24,"publisher":"Springer Science and Business Media LLC","issue":"1","license":[{"start":{"date-parts":[[2022,1,3]],"date-time":"2022-01-03T00:00:00Z","timestamp":1641168000000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"},{"start":{"date-parts":[[2022,1,3]],"date-time":"2022-01-03T00:00:00Z","timestamp":1641168000000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"}],"content-domain":{"domain":["link.springer.com"],"crossmark-restriction":false},"short-container-title":["BMC Pediatr"],"published-print":{"date-parts":[[2022,12]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Background<\/jats:title>\n                    <jats:p>Langerhans cell histiocytosis (LCH) is a rare condition that has a variety of clinical manifestations. But LCH in children localized only in the hepatobiliary system is unusual.<\/jats:p>\n                    <jats:p>Case presentation.<\/jats:p>\n                    <jats:p>\n                      Here we reported a rare case of a 2-year-old boy who was serendipitously found to have elevated liver enzymes while undergoing treatment of a perianal abscess. After a period of earlier conservative treatment in another hospital, the perianal abscess had resolved but the levels of liver enzymes were still rising slowly. The child was then referred to our institution for a definitive diagnosis. After laboratory tests, imaging and pathological examinations, a diagnosis of liver cirrhosis and sclerosing cholangitis was established, although the cause was unclear. Subsequently, living-donor liver transplantation was performed due to deterioration in liver function. Following successful liver transplantation, a diagnosis of LCH localized only within the hepatobiliary system was finally confirmed, based on additional pathological and imaging investigation. Additionally, the\n                      <jats:italic>BRAF V600E<\/jats:italic>\n                      mutation in this patient was also confirmed. The child has now recovered without evidence of LCH recurrence.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusions<\/jats:title>\n                    <jats:p>LCH localized only within the hepatobiliary system is unusual. The presence of unexplainable sclerosing cholangitis and liver cirrhosis in any child should raise the suspicion of LCH.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1186\/s12887-021-03090-4","type":"journal-article","created":{"date-parts":[[2022,1,3]],"date-time":"2022-01-03T04:05:28Z","timestamp":1641182728000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":7,"title":["Liver transplantation in a child with liver cirrhosis caused by langerhans cell histiocytosis: a case report"],"prefix":"10.1186","volume":"22","author":[{"given":"Qi","family":"Wang","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Shuguang","family":"Jin","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Bo","family":"Xiang","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-5540-0878","authenticated-orcid":false,"given":"Jing","family":"Chen","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2022,1,3]]},"reference":[{"issue":"2","key":"3090_CR1","doi-asserted-by":"publisher","first-page":"175","DOI":"10.1002\/pbc.24367","volume":"60","author":"R Haupt","year":"2013","unstructured":"Haupt R, Minkov M, Astigarraga I, Sch\u00e4fer E, Nanduri V, Jubran R, Egeler RM, Janka G, Micic D, Rodriguez-Galindo C, et al. Langerhans cell histiocytosis (LCH): guidelines for diagnosis, clinical work-up, and treatment for patients till the age of 18 years. Pediatr Blood Cancer. 2013;60(2):175\u201384.","journal-title":"Pediatr Blood Cancer"},{"issue":"1","key":"3090_CR2","doi-asserted-by":"publisher","first-page":"74","DOI":"10.1007\/s12185-013-1364-2","volume":"98","author":"K Horibe","year":"2013","unstructured":"Horibe K, Saito AM, Takimoto T, Tsuchida M, Manabe A, Shima M, Ohara A, Mizutani S. Incidence and survival rates of hematological malignancies in Japanese children and adolescents (2006\u20132010): based on registry data from the Japanese Society of Pediatric Hematology. Int J Hematol. 2013;98(1):74\u201388.","journal-title":"Int J Hematol"},{"issue":"1","key":"3090_CR3","doi-asserted-by":"publisher","first-page":"6","DOI":"10.5114\/pdia.2017.67095","volume":"35","author":"M Jezierska","year":"2018","unstructured":"Jezierska M, Stefanowicz J, Romanowicz G, Kosiak W, Lange M. 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