{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,2,5]],"date-time":"2026-02-05T18:44:36Z","timestamp":1770317076412,"version":"3.49.0"},"reference-count":17,"publisher":"BMJ","issue":"2","content-domain":{"domain":["bmj.com"],"crossmark-restriction":true},"short-container-title":["BMJ Case Rep"],"accepted":{"date-parts":[[2026,1,7]]},"published-print":{"date-parts":[[2026,2]]},"abstract":"<jats:p>Langerhans cell histiocytosis (LCH) is a rare clonal myeloid neoplasm with varied and often organ-specific manifestations. Hepatic involvement in adult LCH is rare and frequently results in delayed diagnosis due to its non-specific clinical and biochemical features. We describe the case of a woman in her late twenties who presented with intermittent upper abdominal discomfort, fatigue and weight loss. Despite extensive imaging, serological workup and initial liver biopsy, no definitive diagnosis could be established. Ultimately, diagnostic laparoscopy and histopathological examination with immunohistochemistry confirmed the diagnosis of multifocal hepatic LCH. This case highlights the importance of considering LCH in the differential diagnosis of unexplained hepatomegaly with cholestatic liver enzyme elevation and systemic symptoms. Early liver biopsy with appropriate immunophenotyping is critical for timely diagnosis and initiation of treatment.<\/jats:p>","DOI":"10.1136\/bcr-2025-267970","type":"journal-article","created":{"date-parts":[[2026,2,4]],"date-time":"2026-02-04T14:56:24Z","timestamp":1770216984000},"page":"e267970","update-policy":"https:\/\/doi.org\/10.1136\/crossmarkpolicy","source":"Crossref","is-referenced-by-count":0,"title":["Uncommon presentation of hepatic Langerhans cell histiocytosis in a young adult: a diagnostic challenge"],"prefix":"10.1136","volume":"19","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-0593-2141","authenticated-orcid":false,"given":"Karthikeyan","family":"Kaliyamurthi","sequence":"first","affiliation":[{"name":"Medical Oncology, Mahatma Gandhi medical college and Research Institute, Sri balaji Vidyapeeth University, Pondicherry, Puducherry, India"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Vishva","family":"Babu","sequence":"additional","affiliation":[{"name":"Medical Oncology, Mahatma Gandhi Medical College and Research Institute, Sri balaji Vidyapeeth university, Pondicherry, Pondicherry, India"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Karthikeyan","family":"Vadivazhagan","sequence":"additional","affiliation":[{"name":"Pathology, Mahatma Gandhi Medical College and Research Institute, Sri Balaji Vidyapeeth university, Pondicherry, Pondicherry, India"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"239","published-online":{"date-parts":[[2026,2,4]]},"reference":[{"key":"2026020406552449000_19.2.e267970.1","doi-asserted-by":"crossref","first-page":"2601","DOI":"10.1182\/blood.2021014343","article-title":"International expert consensus recommendations for the diagnosis and treatment of Langerhans cell histiocytosis in adults","volume":"139","author":"Goyal","year":"2022","journal-title":"Blood"},{"key":"2026020406552449000_19.2.e267970.2","unstructured":"Lebbe C , et al . n.d. Hepatic LCH in adults: Data from the French registry."},{"key":"2026020406552449000_19.2.e267970.3","first-page":"e83","article-title":"LCH in adults: Clinical and molecular insights","volume":"22","author":"Rodr\u00edguez-Galindo","year":"2011","journal-title":"Eur J Intern Med"},{"key":"2026020406552449000_19.2.e267970.4","first-page":"1268","article-title":"International expert consensus recommendations for adult LCH","volume":"103","author":"Emile","year":"2018","journal-title":"Haematologica"},{"key":"2026020406552449000_19.2.e267970.5","first-page":"2601","article-title":"Multisystem LCH presenting with liver dysfunction: A literature review","volume":"139","author":"Chang","year":"2022","journal-title":"Blood"},{"key":"2026020406552449000_19.2.e267970.6","first-page":"56","article-title":"Radiology Editorial Board","volume":"12","year":"2024","journal-title":"Biomed Rep"},{"key":"2026020406552449000_19.2.e267970.7","first-page":"751","article-title":"PET-CT characteristics of hepatic LCH","volume":"241","author":"Bhuvana","year":"2024","journal-title":"Radiology"},{"key":"2026020406552449000_19.2.e267970.8","first-page":"34","article-title":"Percutaneous biopsy pitfalls in hepatic Langerhans LCH: a case series","volume":"27","author":"Lee","year":"2024","journal-title":"Nucl Med Rev"},{"key":"2026020406552449000_19.2.e267970.9","first-page":"1520","article-title":"Blind biopsy miss rates in adult hepatic LCH","volume":"21","author":"Hanley","year":"2023","journal-title":"Clin Gastroenterol Hepatol"},{"key":"2026020406552449000_19.2.e267970.10","first-page":"482","article-title":"Surgical biopsy and immunophenotyping in hepatic LCH","volume":"65","author":"Zhang","year":"2024","journal-title":"Histopathology"},{"key":"2026020406552449000_19.2.e267970.11","first-page":"487","article-title":"Molecular pathogenesis of LCH: BRAF and MAP2K1 mutations","volume":"69","author":"Haroche","year":"2016","journal-title":"J Clin Pathol"},{"key":"2026020406552449000_19.2.e267970.12","first-page":"1230","article-title":"Vemurafenib in refractory LCH: phase II trial results","volume":"126","author":"Haroche","year":"2015","journal-title":"Blood"},{"key":"2026020406552449000_19.2.e267970.13","first-page":"e31","article-title":"Prognosis and outcomes of adult hepatic LCH: a registry analysis","volume":"185","author":"Abdallah","year":"2019","journal-title":"Br J Haematol"},{"key":"2026020406552449000_19.2.e267970.14","doi-asserted-by":"crossref","first-page":"335","DOI":"10.5306\/wjco.v12.i5.335","article-title":"Hepatic Langerhans cell histiocytosis: A review","volume":"12","author":"Fu","year":"2021","journal-title":"World J Clin Oncol"},{"key":"2026020406552449000_19.2.e267970.15","doi-asserted-by":"crossref","first-page":"e1347","DOI":"10.1093\/oncolo\/oyae175","article-title":"Liver involvement with Langerhans cell histiocytosis in adults","volume":"29","author":"Chang","year":"2024","journal-title":"Oncologist"},{"key":"2026020406552449000_19.2.e267970.16","first-page":"2601","article-title":"Multidisciplinary management of adult LCH: experience from a single centre","volume":"139","author":"Hanley","year":"2022","journal-title":"Blood"},{"key":"2026020406552449000_19.2.e267970.17","first-page":"509","article-title":"Revised classification of histiocytic disorders","volume":"75","author":"Aric\u00f2","year":"2022","journal-title":"Hepatology"}],"container-title":["BMJ Case Reports"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/syndication.highwire.org\/content\/doi\/10.1136\/bcr-2025-267970","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,2,4]],"date-time":"2026-02-04T14:56:27Z","timestamp":1770216987000},"score":1,"resource":{"primary":{"URL":"https:\/\/casereports.bmj.com\/lookup\/doi\/10.1136\/bcr-2025-267970"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026,2]]},"references-count":17,"journal-issue":{"issue":"2","published-online":{"date-parts":[[2026,2,4]]},"published-print":{"date-parts":[[2026,2]]}},"alternative-id":["10.1136\/bcr-2025-267970"],"URL":"https:\/\/doi.org\/10.1136\/bcr-2025-267970","relation":{},"ISSN":["1757-790X"],"issn-type":[{"value":"1757-790X","type":"electronic"}],"subject":[],"published":{"date-parts":[[2026,2]]}}}