{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,2,10]],"date-time":"2026-02-10T03:51:34Z","timestamp":1770695494053,"version":"3.49.0"},"reference-count":15,"publisher":"Wiley","issue":"1","license":[{"start":{"date-parts":[[2026,2,9]],"date-time":"2026-02-09T00:00:00Z","timestamp":1770595200000},"content-version":"vor","delay-in-days":39,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"},{"start":{"date-parts":[[2026,1,1]],"date-time":"2026-01-01T00:00:00Z","timestamp":1767225600000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/doi.wiley.com\/10.1002\/tdm_license_1.1"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Case Reports in Hematology"],"published-print":{"date-parts":[[2026,1]]},"abstract":"<jats:p>\n                    We report a case of hemophagocytic lymphohistiocytosis (HLH) secondary to disseminated Bacillus Calmette\u2013Gu\u00e9rin (BCG) infection in an 80\u2010year\u2010old man treated with intravesical BCG for non\u2013muscle\u2010invasive bladder cancer. The patient presented with several weeks of constitutional symptoms including night sweats, fatigue, weight loss, confusion, and pancytopenia. Laboratory studies revealed profound inflammation, coagulopathy, and hepatocellular injury. HLH was suspected clinically and supported by a high H\u2010score. Extensive infectious and autoimmune workup was negative.\n                    <jats:italic>Mycobacterium bovis<\/jats:italic>\n                    was subsequently isolated from urine and bone marrow cultures, which confirmed a diagnosis of disseminated BCG infection.\n                  <\/jats:p>\n                  <jats:p>\n                    The patient was treated with a combination of antimycobacterial therapy (isoniazid, rifampin, and ethambutol), intravenous immunoglobulin (IVIG), corticosteroids, and anakinra. Anakinra, an interleukin\u20101 receptor antagonist, was used both to minimize high\u2010dose steroids, given the protracted inflammatory response anticipated with\n                    <jats:italic>Mycobacterium<\/jats:italic>\n                    infection, and to prevent steroid\u2010related complications related to cardiac surgery. This therapeutic strategy was associated with improvement of cytopenias, normalization of inflammatory markers, and gradual clinical recovery. This case highlights the rare but serious complication of HLH triggered by disseminated BCG and suggests that targeted immunomodulation with anakinra may be a useful adjunct in selected, complex presentations of infection\u2010associated HLH.\n                  <\/jats:p>","DOI":"10.1155\/crh\/5761690","type":"journal-article","created":{"date-parts":[[2026,2,9]],"date-time":"2026-02-09T10:49:13Z","timestamp":1770634153000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["Hemophagocytic Lymphohistiocytosis Secondary to Disseminated Bacillus Calmette\u2013Gu\u00e9rin (BCG): A Steroid\u2010Sparing Strategy in a Patient Awaiting Cardiac Surgery\u2014A Case Report"],"prefix":"10.1155","volume":"2026","author":[{"ORCID":"https:\/\/orcid.org\/0009-0003-9999-258X","authenticated-orcid":false,"given":"Kaitlin","family":"Quinlan","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-4116-6376","authenticated-orcid":false,"given":"Tyler","family":"MacDonald","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-2837-1973","authenticated-orcid":false,"given":"Terence C.","family":"Wuerz","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-9455-6138","authenticated-orcid":false,"given":"Ryan","family":"Zarychanski","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"311","published-online":{"date-parts":[[2026,2,9]]},"reference":[{"key":"e_1_2_10_1_2","doi-asserted-by":"publisher","DOI":"10.1002\/art.38690"},{"key":"e_1_2_10_2_2","doi-asserted-by":"publisher","DOI":"10.1016\/S0140-6736(13)61048-X"},{"key":"e_1_2_10_3_2","doi-asserted-by":"publisher","DOI":"10.1182\/blood.2018894618"},{"key":"e_1_2_10_4_2","doi-asserted-by":"publisher","DOI":"10.1016\/s0002-9343(02)01066-5"},{"key":"e_1_2_10_5_2","doi-asserted-by":"publisher","DOI":"10.5489\/cuaj.1411"},{"key":"e_1_2_10_6_2","doi-asserted-by":"publisher","DOI":"10.12890\/2022_003395"},{"key":"e_1_2_10_7_2","doi-asserted-by":"publisher","DOI":"10.1001\/jama.2020.17598"},{"key":"e_1_2_10_8_2","doi-asserted-by":"publisher","DOI":"10.3389\/fimmu.2021.676132"},{"key":"e_1_2_10_9_2","doi-asserted-by":"publisher","DOI":"10.3109\/21681805.2013.836724"},{"key":"e_1_2_10_10_2","doi-asserted-by":"publisher","DOI":"10.1016\/s0399-8320(06)73208-0"},{"key":"e_1_2_10_11_2","doi-asserted-by":"crossref","unstructured":"Stojanovicet al. Severe Hemophagocytic Syndrome After Intravesical BCG Instillation with a Fatal Outcome 2022 https:\/\/www.researchgate.net\/publication\/361459563_Severe_hemophagocytic_syndrome_after_intravesical_BCG_instillation_with_a_fatal_outcome.","DOI":"10.2298\/VSP210412065S"},{"key":"e_1_2_10_12_2","first-page":"531","article-title":"Safety Profile of Anakinra in the Management of Rheumatologic, Metabolic and Autoinflammatory Disorders","volume":"34","author":"Lopalco G.","year":"2016","journal-title":"Clinical & Experimental Rheumatology"},{"key":"e_1_2_10_13_2","doi-asserted-by":"publisher","DOI":"10.1136\/annrheumdis-2015-eular.2232"},{"key":"e_1_2_10_14_2","unstructured":"Getting It Right First Time [GIRFT] Haemophagocytic Lymphohistiocytosis (HLH) Guide: Final Version v1.3 June 2025 2025 https:\/\/gettingitrightfirsttime.co.uk\/wp-content\/uploads\/2025\/06\/HLH-Guide-final-version-v1.3-June-2025.pdf."},{"key":"e_1_2_10_15_2","doi-asserted-by":"publisher","DOI":"10.12890\/2023_004009"}],"container-title":["Case Reports in Hematology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1155\/crh\/5761690","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/full-xml\/10.1155\/crh\/5761690","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1155\/crh\/5761690","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,2,9]],"date-time":"2026-02-09T10:49:16Z","timestamp":1770634156000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1155\/crh\/5761690"}},"subtitle":[],"editor":[{"given":"Yoshito","family":"Nishimura","sequence":"additional","affiliation":[],"role":[{"role":"editor","vocabulary":"crossref"}]}],"short-title":[],"issued":{"date-parts":[[2026,1]]},"references-count":15,"journal-issue":{"issue":"1","published-print":{"date-parts":[[2026,1]]}},"alternative-id":["10.1155\/crh\/5761690"],"URL":"https:\/\/doi.org\/10.1155\/crh\/5761690","archive":["Portico"],"relation":{},"ISSN":["2090-6560","2090-6579"],"issn-type":[{"value":"2090-6560","type":"print"},{"value":"2090-6579","type":"electronic"}],"subject":[],"published":{"date-parts":[[2026,1]]},"assertion":[{"value":"2025-09-24","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2025-12-22","order":2,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2026-02-09","order":3,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}],"article-number":"5761690"}}