{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,19]],"date-time":"2026-10-19T10:46:29Z","timestamp":1792406789541,"version":"4.4.0"},"reference-count":32,"publisher":"Wiley","issue":"5","license":[{"start":{"date-parts":[[2026,8,16]],"date-time":"2026-08-16T00:00:00Z","timestamp":1786838400000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"},{"start":{"date-parts":[[2026,8,16]],"date-time":"2026-08-16T00:00:00Z","timestamp":1786838400000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/doi.wiley.com\/10.1002\/tdm_license_1.1"}],"funder":[{"DOI":"10.13039\/100014823","name":"VGH and UBC Hospital Foundation","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100014823","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Hematological Oncology"],"published-print":{"date-parts":[[2026,9]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n                  <jats:p>\n                    Langerhans cell histiocytosis (LCH) is a rare clonal myeloid neoplasm. Canadian data on clinical characteristics, molecular profile, and treatment outcomes is limited. This study aims to report the initial experience of a Canadian rare diseases program, reflecting \u201creal\u2010world\u201d diagnostic pathways, referral patterns, and treatment heterogeneity across multiple provinces. We conducted a retrospective review of patients managed in an adult\u2010care cohort with histologically confirmed LCH diagnosed between 2000 and 2025 across multiple Canadian centers. Patients diagnosed as children and subsequently transferred to adult care were included. Clinical features, radiologic findings, histopathology, molecular testing, treatment approaches, and outcomes were collected and analyzed. Thirty\u2010one patients were identified, with a median age at diagnosis of 42\u00a0years (range: 2\u201384) and a male predominance (65%). Bone (74%), lung (29%), and skin (16%) were the most commonly involved sites. Concomitant or subsequent malignancies were present in 19% of patients. Molecular testing found BRAFV600E mutations in 44% of tested patients. The most common first\u2010line systemic therapy was cytarabine (\n                    <jats:italic>n<\/jats:italic>\n                    \u00a0=\u00a010), followed by other drugs such as hydroxyurea, vemurafenib, and cladribine. This series represents the initial experience of a Canadian rare disease referral program and captures the clinical heterogeneity and longitudinal adult care of patients with LCH across multiple provinces. Variability in treatment approaches highlights the need for collaborative prospective natural history studies and coordinated clinical trials.\n                  <\/jats:p>","DOI":"10.1002\/hon.70241","type":"journal-article","created":{"date-parts":[[2026,8,17]],"date-time":"2026-08-17T06:23:35Z","timestamp":1786947815000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["Langerhans Cell Histiocytosis in Adults: A Canadian Multicenter Case Series"],"prefix":"10.1002","volume":"44","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-9188-8380","authenticated-orcid":false,"given":"Stephanie","family":"Quon","sequence":"first","affiliation":[{"name":"Faculty of Medicine University of British Columbia  Vancouver British Columbia Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yaswanta","family":"Gummadi","sequence":"additional","affiliation":[{"name":"Department of Medicine Dalhousie University  Halifax Nova Scotia Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-2059-6895","authenticated-orcid":false,"given":"Ibrahim","family":"Elsharawi","sequence":"additional","affiliation":[{"name":"Department of Pathology Dalhousie University  Halifax Nova Scotia Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jessica","family":"Dobson","sequence":"additional","affiliation":[{"name":"Department of Diagnostic Radiology Dalhousie University  Halifax Nova Scotia Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-9181-379X","authenticated-orcid":false,"given":"Mariam","family":"Goubran","sequence":"additional","affiliation":[{"name":"Division of Hematology University of British Columbia  Vancouver British Columbia Canada"},{"name":"Coastal Rare Inflammatory Diseases Program  Halifax Nova Scotia Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-4718-5212","authenticated-orcid":false,"given":"Ryan J.","family":"Stubbins","sequence":"additional","affiliation":[{"name":"Division of Hematology University of British Columbia  Vancouver British Columbia Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Eli L.","family":"Diamond","sequence":"additional","affiliation":[{"name":"Department of Neurology Memorial Sloan Kettering Cancer Center  New York New York USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-9551-2951","authenticated-orcid":false,"given":"Luke Y. C.","family":"Chen","sequence":"additional","affiliation":[{"name":"Division of Hematology University of British Columbia  Vancouver British Columbia Canada"},{"name":"Coastal Rare Inflammatory Diseases Program  Halifax Nova Scotia Canada"},{"name":"Division of Hematology Dalhousie University  Halifax Nova Scotia 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