{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,1,17]],"date-time":"2026-01-17T23:42:29Z","timestamp":1768693349956,"version":"3.49.0"},"reference-count":26,"publisher":"BMJ","issue":"2","content-domain":{"domain":["bmj.com"],"crossmark-restriction":true},"short-container-title":["BMJ Case Rep"],"accepted":{"date-parts":[[2019,1,28]]},"published-print":{"date-parts":[[2019,2]]},"abstract":"<jats:p>A 27-year-old man presented with high-grade intermittent fever for 4 months, generalised fatigue for 2 months, intermittent gum bleeds for 1\u2009month and loss of weight of 15 kg. He appeared cachectic with generalised wasting, had pallor and features of reticuloendothelial system proliferation. His liver span was 17\u2009cm. He had massive splenomegaly. His cardiovascular, respiratory and neurological examination were normal. He was diagnosed to have visceral leishmaniasis (VL) based on bone marrow (BM) examination that showed Leishmania donovani (LD) bodies and was treated with liposomal amphotericin (LA). During the course of therapy, he developed bleeding from various mucosal and venepuncture sites. His further evaluation, which included a repeat BM aspirate, showed haemophagocytes. Final diagnosis made was VL with secondary haemophagocytic lymphohistiocytosis. He was continued on LA with intravenous hydrocortisone. He developed refractory distributive shock with multiorgan dysfunction and succumbed to his illness.<\/jats:p>","DOI":"10.1136\/bcr-2018-226361","type":"journal-article","created":{"date-parts":[[2019,2,14]],"date-time":"2019-02-14T04:56:13Z","timestamp":1550120173000},"page":"e226361","update-policy":"https:\/\/doi.org\/10.1136\/crossmarkpolicy","source":"Crossref","is-referenced-by-count":2,"title":["Visceral leishmaniasis with haemophagocytic lymphohistiocytosis"],"prefix":"10.1136","volume":"12","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-5405-8289","authenticated-orcid":false,"given":"John Titus","family":"George","sequence":"first","affiliation":[{"name":"Department of General Medicine, Christian Medical College and Hospital, Vellore, Tamil Nadu, India"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Mohammad","family":"Sadiq","sequence":"additional","affiliation":[{"name":"Department of General Medicine, Christian Medical College and Hospital, Vellore, Tamil Nadu, India"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Elanthenral","family":"Sigamani","sequence":"additional","affiliation":[{"name":"Department of Pathology, Christian Medical College and Hospital, Vellore, Tamil Nadu, India"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Alice Joan","family":"Mathuram","sequence":"additional","affiliation":[{"name":"Department of General Medicine, Christian Medical College and Hospital, Vellore, Tamil Nadu, India"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"239","published-online":{"date-parts":[[2019,2,13]]},"reference":[{"key":"2025082712123606000_12.2.e226361.1","unstructured":"WHO | Leishmaniasis [Internet]. WHO. http:\/\/www.who.int\/mediacentre\/factsheets\/fs375\/en\/ (cited 9 Nov 2017)."},{"key":"2025082712123606000_12.2.e226361.2","doi-asserted-by":"publisher","DOI":"10.1016\/j.bbmt.2009.11.014"},{"key":"2025082712123606000_12.2.e226361.3","doi-asserted-by":"publisher","DOI":"10.1002\/pbc.21039"},{"key":"2025082712123606000_12.2.e226361.4","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pntd.0000114"},{"key":"2025082712123606000_12.2.e226361.5","doi-asserted-by":"crossref","DOI":"10.1136\/bmjopen-2014-005408","article-title":"Visceral leishmaniasis-associated mortality in Bangladesh: a retrospective cross-sectional study","volume":"4","author":"Huda","year":"2014","journal-title":"BMJ Open"},{"key":"2025082712123606000_12.2.e226361.6","unstructured":"Organization WH, UNICEF. Annual report 2016: intervention and implementation research: World Health Organization, 2017."},{"key":"2025082712123606000_12.2.e226361.7","doi-asserted-by":"publisher","DOI":"10.1111\/j.1462-5822.2008.01146.x"},{"key":"2025082712123606000_12.2.e226361.8","unstructured":"Mandell GL , Bennett JE , Dolin R , eds. Mandell, Douglas, and Bennett\u2019s principles and practice of infectious diseases. 7th ed. Philadelphia, PA: Churchill Livingstone\/Elsevier, 2010:2."},{"key":"2025082712123606000_12.2.e226361.9","doi-asserted-by":"publisher","DOI":"10.1016\/j.jbspin.2011.10.015"},{"key":"2025082712123606000_12.2.e226361.10","doi-asserted-by":"publisher","DOI":"10.1182\/asheducation-2015.1.183"},{"key":"2025082712123606000_12.2.e226361.11","doi-asserted-by":"crossref","first-page":"1503","DOI":"10.1016\/S0140-6736(13)61048-X","article-title":"Adult haemophagocytic syndrome","volume":"383","author":"Ramos-Casals","year":"2014","journal-title":"The Lancet"},{"key":"2025082712123606000_12.2.e226361.12","doi-asserted-by":"crossref","first-page":"121","DOI":"10.1093\/omcr\/omw043","article-title":"Dengue fever complicated by hemophagocytosis","volume":"2016","author":"Koshy","year":"2016","journal-title":"Oxf Med Case Reports"},{"key":"2025082712123606000_12.2.e226361.13","doi-asserted-by":"publisher","DOI":"10.1016\/S1473-3099(07)70290-6"},{"key":"2025082712123606000_12.2.e226361.14","doi-asserted-by":"crossref","first-page":"203","DOI":"10.1111\/bjh.14147","article-title":"Advances in the pathogenesis of primary and secondary haemophagocytic lymphohistiocytosis: differences and similarities","volume":"174","author":"Brisse","year":"2016","journal-title":"Br J Haematol"},{"key":"2025082712123606000_12.2.e226361.15","doi-asserted-by":"publisher","DOI":"10.1182\/blood-2011-08-374199"},{"key":"2025082712123606000_12.2.e226361.16","doi-asserted-by":"publisher","DOI":"10.1002\/art.20198"},{"key":"2025082712123606000_12.2.e226361.17","doi-asserted-by":"crossref","first-page":"387","DOI":"10.1007\/s00281-015-0493-5","article-title":"New monogenic autoinflammatory diseases-a clinical overview","volume":"37","author":"Canna","year":"2015","journal-title":"Semin Immunopathol"},{"key":"2025082712123606000_12.2.e226361.18","doi-asserted-by":"crossref","first-page":"35","DOI":"10.1016\/j.cytogfr.2014.05.005","article-title":"Cytokine balance and cytokine-driven natural killer cell dysfunction in systemic juvenile idiopathic arthritis","volume":"26","author":"Avau","year":"2015","journal-title":"Cytokine Growth Factor Rev"},{"key":"2025082712123606000_12.2.e226361.19","doi-asserted-by":"publisher","DOI":"10.4049\/jimmunol.176.8.4530"},{"key":"2025082712123606000_12.2.e226361.20","doi-asserted-by":"crossref","first-page":"436","DOI":"10.1128\/JVI.72.1.436-441.1998","article-title":"Herpes simplex virus type 1 renders infected cells resistant to cytotoxic T-lymphocyte-induced apoptosis","volume":"72","author":"Jerome","year":"1998","journal-title":"J Virol"},{"key":"2025082712123606000_12.2.e226361.21","doi-asserted-by":"publisher","DOI":"10.1182\/blood-2005-04-1406"},{"key":"2025082712123606000_12.2.e226361.22","doi-asserted-by":"publisher","DOI":"10.1542\/peds.106.4.e58"},{"key":"2025082712123606000_12.2.e226361.23","doi-asserted-by":"publisher","DOI":"10.3201\/eid0606.000608"},{"key":"2025082712123606000_12.2.e226361.24","doi-asserted-by":"crossref","first-page":"69","DOI":"10.2147\/JBM.S46255","article-title":"Hemophagocytic lymphohistiocytosis: review of etiologies and management","volume":"5","author":"George","year":"2014","journal-title":"J Blood Med"},{"key":"2025082712123606000_12.2.e226361.25","doi-asserted-by":"publisher","DOI":"10.1016\/j.jinf.2008.02.013"},{"key":"2025082712123606000_12.2.e226361.26","doi-asserted-by":"crossref","first-page":"e6935","DOI":"10.1097\/MD.0000000000006935","article-title":"Clinical features and prognostic factors of adult secondary hemophagocytic syndrome: Analysis of 47 cases","volume":"96","author":"Guo","year":"2017","journal-title":"Medicine"}],"container-title":["BMJ Case Reports"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/syndication.highwire.org\/content\/doi\/10.1136\/bcr-2018-226361","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,8,27]],"date-time":"2025-08-27T19:12:43Z","timestamp":1756321963000},"score":1,"resource":{"primary":{"URL":"https:\/\/casereports.bmj.com\/lookup\/doi\/10.1136\/bcr-2018-226361"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2019,2]]},"references-count":26,"journal-issue":{"issue":"2","published-online":{"date-parts":[[2019,2,13]]},"published-print":{"date-parts":[[2019,2]]}},"alternative-id":["10.1136\/bcr-2018-226361"],"URL":"https:\/\/doi.org\/10.1136\/bcr-2018-226361","relation":{},"ISSN":["1757-790X"],"issn-type":[{"value":"1757-790X","type":"electronic"}],"subject":[],"published":{"date-parts":[[2019,2]]}}}