{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,29]],"date-time":"2026-07-29T05:58:11Z","timestamp":1785304691243,"version":"3.55.0"},"reference-count":17,"publisher":"Wiley","issue":"1","license":[{"start":{"date-parts":[[2026,7,28]],"date-time":"2026-07-28T00:00:00Z","timestamp":1785196800000},"content-version":"vor","delay-in-days":208,"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 Gastrointestinal Medicine"],"published-print":{"date-parts":[[2026,1]]},"abstract":"<jats:sec>\n                    <jats:title>Background<\/jats:title>\n                    <jats:p>Hemophagocytic lymphohistiocytosis (HLH) is a rare yet life\u2010threatening hyperinflammatory syndrome, increasingly reported in patients with inflammatory bowel disease (IBD) receiving biologic therapy. While Epstein\u2013Barr virus and cytomegalovirus represent the most frequent infectious triggers, visceral leishmaniasis (VL) is extremely uncommon and may be easily ignored outside endemic areas.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Case Presentation<\/jats:title>\n                    <jats:p>\n                      We report the case of a 31\u2010year\u2010old man with Crohn\u2019s disease treated with escalated anti\u2010TNF\u03b1 therapy who presented with persistent fever, pancytopenia, hyperferritinemia, hypertriglyceridemia, and progressive systemic inflammation. Both infectious and oncologic workups were initially inconclusive. PET\/CT showed splenomegaly, diffuse splenic, and bone marrow hypermetabolism, raising suspicion for a lymphoproliferative disorder; however, bone marrow aspirate revealed systemic inflammation and hemophagocytosis, suggesting HLH and detected intracellular amastigotes consistent with\n                      <jats:italic>Leishmania<\/jats:italic>\n                      spp., subsequently confirmed by serology. Liposomal amphotericin B prompted rapid clinical and biochemical improvement, with complete resolution of HLH. Anti\u2010TNF\u03b1 was discontinued and replaced with anti\u2013IL\u201012\/23 therapy to maintain IBD control while minimizing infectious risk.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusion<\/jats:title>\n                    <jats:p>\n                      In patients receiving anti\u2010TNF therapy who present with persistent fever, cytopenias, hyperferritinemia, and unexplained systemic inflammation, HLH should be promptly investigated. Early bone marrow examination may facilitate diagnosis by revealing both hemophagocytosis and\n                      <jats:italic>Leishmania<\/jats:italic>\n                      amastigotes, enabling timely treatment. Following resolution of infection, transition to a biologic with a different mechanism of action, such as anti\u2013IL\u201012\/23 therapy, may represent a reasonable strategy for long\u2010term IBD management.\n                    <\/jats:p>\n                  <\/jats:sec>","DOI":"10.1155\/crgm\/6403158","type":"journal-article","created":{"date-parts":[[2026,7,29]],"date-time":"2026-07-29T04:58:11Z","timestamp":1785301091000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["Visceral Leishmaniasis\u2013Associated Hemophagocytic Lymphohistiocytosis in a Patient With Crohn\u2019s Disease in Therapy With Anti\u2010TNF\u03b1: A Challenging Diagnostic Case Report"],"prefix":"10.1155","volume":"2026","author":[{"ORCID":"https:\/\/orcid.org\/0009-0001-5097-0174","authenticated-orcid":false,"given":"Caterina","family":"Pelligra","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0008-9097-6379","authenticated-orcid":false,"given":"Lorenzo","family":"Bolis","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-4345-6051","authenticated-orcid":false,"given":"Andrea","family":"Costantino","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-1558-8604","authenticated-orcid":false,"given":"Maurizio","family":"Vecchi","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2026,7,28]]},"reference":[{"key":"e_1_2_12_1_2","doi-asserted-by":"publisher","DOI":"10.3389\/fimmu.2023.1210041"},{"key":"e_1_2_12_2_2","doi-asserted-by":"publisher","DOI":"10.1016\/S2665-9913(23)00287-3"},{"key":"e_1_2_12_3_2","doi-asserted-by":"publisher","DOI":"10.1016\/j.berh.2020.101515"},{"key":"e_1_2_12_4_2","doi-asserted-by":"publisher","DOI":"10.1007\/s10620-020-06157-1"},{"key":"e_1_2_12_5_2","doi-asserted-by":"publisher","DOI":"10.3389\/fimmu.2025.1575297"},{"key":"e_1_2_12_6_2","doi-asserted-by":"publisher","DOI":"10.1111\/apa.15966"},{"key":"e_1_2_12_7_2","doi-asserted-by":"publisher","DOI":"10.1016\/j.semarthrit.2015.07.003"},{"key":"e_1_2_12_8_2","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pntd.0009944"},{"key":"e_1_2_12_9_2","doi-asserted-by":"publisher","DOI":"10.1097\/MPH.0b013e318286d619"},{"key":"e_1_2_12_10_2","first-page":"31","article-title":"Hemophagocytic Lymphohistiocytosis Mimics Many Common Conditions: Case Series and Review of Literature","volume":"49","author":"Akenroye A. T.","year":"2017","journal-title":"European Annals of Allergy and Clinical Immunology"},{"key":"e_1_2_12_11_2","doi-asserted-by":"publisher","DOI":"10.4269\/ajtmh.22-0302"},{"key":"e_1_2_12_12_2","doi-asserted-by":"publisher","DOI":"10.1080\/03009742.2018.1542142"},{"key":"e_1_2_12_13_2","first-page":"503","article-title":"Visceral Leishmaniasis and Anti\u2013TNF-\u03b1 Therapy: Case Report and Review of the Literature","volume":"27","author":"De Leonardis F.","year":"2009","journal-title":"Clinical & Experimental Rheumatology"},{"key":"e_1_2_12_14_2","doi-asserted-by":"publisher","DOI":"10.1016\/j.jaci.2022.06.017"},{"key":"e_1_2_12_15_2","doi-asserted-by":"publisher","DOI":"10.3390\/tropicalmed8050276"},{"key":"e_1_2_12_16_2","doi-asserted-by":"publisher","DOI":"10.1016\/j.idcr.2024.e02059"},{"key":"e_1_2_12_17_2","doi-asserted-by":"publisher","DOI":"10.1002\/ccr3.71351"}],"container-title":["Case Reports in Gastrointestinal Medicine"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1155\/crgm\/6403158","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/full-xml\/10.1155\/crgm\/6403158","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1155\/crgm\/6403158","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,7,29]],"date-time":"2026-07-29T04:58:13Z","timestamp":1785301093000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1155\/crgm\/6403158"}},"subtitle":[],"editor":[{"given":"Neetu","family":"Rawal","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"editor"}]}],"short-title":[],"issued":{"date-parts":[[2026,1]]},"references-count":17,"journal-issue":{"issue":"1","published-print":{"date-parts":[[2026,1]]}},"alternative-id":["10.1155\/crgm\/6403158"],"URL":"https:\/\/doi.org\/10.1155\/crgm\/6403158","archive":["Portico"],"relation":{},"ISSN":["2090-6528","2090-6536"],"issn-type":[{"value":"2090-6528","type":"print"},{"value":"2090-6536","type":"electronic"}],"subject":[],"published":{"date-parts":[[2026,1]]},"assertion":[{"value":"2026-01-30","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2026-07-01","order":2,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2026-07-28","order":3,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}],"article-number":"6403158"}}