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Severe cases may progress to liver failure, requiring transplantation. Liver involvement occurs in about one-third of patients with systemic juvenile xanthogranuloma (JXG) and 20% to 60% of pediatric patients with Langerhans cell histiocytosis (LCH), particularly in multiorgan disease. Tyrosine kinase inhibitors show promise in LCH treatment, but optimal timing for treatment cessation remains uncertain. We present 2 pediatric cases, 1 with LCH, and the other with disseminated JXG, along with a literature review emphasizing liver histopathology and transplant considerations. These cases highlight distinct histological patterns. In LCH, progressive bile duct destruction led to ductopenic cholestatic cirrhosis and secondary sclerosing cholangitis. In contrast, in the case of JXG, bile ducts remained intact despite being surrounded by histiocytes. In both, disease localization to larger, segmental portal tracts may reduce liver biopsy sensitivity. In LCH, BRAF inhibitor therapy triggered a granulomatous reaction that could mimic disease recurrence in the liver graft. Other histiocytoses typically spare the bile ducts and do not cause biliary cirrhosis. Recognizing these distinct infiltration patterns can aid diagnosis and management.<\/jats:p>","DOI":"10.1177\/10935266251385405","type":"journal-article","created":{"date-parts":[[2025,10,31]],"date-time":"2025-10-31T11:27:15Z","timestamp":1761910035000},"page":"38-50","update-policy":"https:\/\/doi.org\/10.1177\/sage-journals-update-policy","source":"Crossref","is-referenced-by-count":0,"title":["Bile Duct Targeting or Preservation: Contrasting Liver Histology in Langerhans Cell Histiocytosis and Disseminated Juvenile Xanthogranuloma"],"prefix":"10.1177","volume":"29","author":[{"given":"Margaux","family":"D\u00e4niker","sequence":"first","affiliation":[{"name":"Faculty of Medicine, University of Geneva, Switzerland"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Fr\u00e9d\u00e9ric","family":"Baleydier","sequence":"additional","affiliation":[{"name":"Unit of Paediatric Oncology and Haematology, Department of Women, Child and Adolescent, Geneva University Hospitals, CANSEARCH Research Platform in Paediatric Oncology, Switzerland"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Nathalie M.","family":"Rock","sequence":"additional","affiliation":[{"name":"Swiss Pediatric Liver Center, Department of Women, Child and Adolescent, Geneva University Hospitals, Switzerland"},{"name":"Pediatric Gastroenterology, Hepatology and Nutrition Unit, Division of Pediatric Specialties, Department of Pediatrics, Gynecology, and Obstetrics, Geneva University Hospitals, Switzerland"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"S\u00e9bastien","family":"Menzinger","sequence":"additional","affiliation":[{"name":"Division of Clinical Pathology, Diagnostic Department, and Division of Dermatology and Venereology, Department of Medicine, Geneva University Hospitals, Switzerland"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Barbara E.","family":"Wildhaber","sequence":"additional","affiliation":[{"name":"Swiss Pediatric Liver Center, Department of Women, Child and Adolescent, Geneva University Hospitals, Switzerland"},{"name":"Division of Child and Adolescent Surgery, Department of Pediatrics, Gynecology, and Obstetrics, Geneva University Hospitals, Switzerland"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Val\u00e9rie A.","family":"McLin","sequence":"additional","affiliation":[{"name":"Swiss Pediatric Liver Center, Department of Women, Child and Adolescent, Geneva University Hospitals, Switzerland"},{"name":"Pediatric Gastroenterology, Hepatology and Nutrition Unit, Division of Pediatric Specialties, Department of Pediatrics, Gynecology, and Obstetrics, Geneva University Hospitals, Switzerland"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-1785-792X","authenticated-orcid":false,"given":"Anne-Laure","family":"Rougemont","sequence":"additional","affiliation":[{"name":"Swiss Pediatric Liver Center, Department of Women, Child and Adolescent, Geneva University Hospitals, Switzerland"},{"name":"Pediatric and Fetoplacental Unit &amp; Clinical Molecular Pathology Unit, Division of Clinical Pathology, Diagnostic Department, Geneva University Hospitals, Switzerland"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"179","published-online":{"date-parts":[[2025,10,31]]},"reference":[{"key":"e_1_3_2_2_2","doi-asserted-by":"publisher","DOI":"10.1182\/blood-2016-01-690636"},{"key":"e_1_3_2_3_2","doi-asserted-by":"publisher","DOI":"10.1007\/s00428-024-04018-w"},{"key":"e_1_3_2_4_2","unstructured":"Picarsic J Jacques TS Bychkov A Calaminici M Narula G Alaggio R et al WHO classification of Tumours Editorial Board. 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