{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,21]],"date-time":"2026-10-21T13:08:59Z","timestamp":1792588139684,"version":"4.5.0"},"reference-count":54,"publisher":"American Society of Tropical Medicine and Hygiene","issue":"6","content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2022,12,14]]},"abstract":"<jats:title>ABSTRACT.<\/jats:title>\n                  <jats:p>Hemophagocytic lymphohistiocytosis (HLH) is a rare and fatal complication of visceral leishmaniasis (VL). To provide a basis for early and correct diagnosis and to improve prognosis in the future, we describe a case series of VL-associated HLH in adults in our center in the past decade after review of all reported cases of adult VL-associated HLH in English through May 2022. In our case series, a total of 111 patients were diagnosed with VL. Among these patients, only six cases were diagnosed with VL-associated HLH. All patients tested positive for serology. Leishmania was detected for the first time by bone marrow aspiration (BMA) in three of the six patients and in the other three patients after three or four BMAs. It took more than 1 month from onset to diagnosis of VL for all the six cases, and the longest time was 6 months. Five of the six patients recovered after receiving sodium stibogluconate. VL-associated HLH is rare but potentially life-threatening in adults and predisposes to early delays in diagnosis. However, diagnostic techniques are not complicated or difficult, so it is more important to consider that it is not recognized by physicians. Although guidelines recommend liposomal amphotericin B as the most effective therapy, our experience suggests that sodium stibogluconate can be an alternative option when liposomal amphotericin B is unavailable or unaffordable.<\/jats:p>","DOI":"10.4269\/ajtmh.22-0361","type":"journal-article","created":{"date-parts":[[2022,11,14]],"date-time":"2022-11-14T08:26:25Z","timestamp":1668414385000},"page":"1203-1209","source":"Crossref","is-referenced-by-count":14,"title":["Case Report: Visceral Leishmaniasis-associated Hemophagocytic Lymphohistiocytosis in Adults: A Case Series and Literature Review"],"prefix":"10.4269","volume":"107","author":[{"given":"Yao","family":"Qin","sequence":"first","affiliation":[{"name":"Center of Infectious Diseases, West China Hospital, Sichuan University, Chengdu, China;"},{"name":"Department of Hematology, West China Hospital, Sichuan University, Chengdu, China;"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Xiaoju","family":"Lv","sequence":"additional","affiliation":[{"name":"Center of Infectious Diseases, West China Hospital, Sichuan University, Chengdu, China;"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Qin","family":"Zheng","sequence":"additional","affiliation":[{"name":"Department of Laboratory Medicine, West China Hospital, Sichuan University, Chengdu, China;"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Qian","family":"Wu","sequence":"additional","affiliation":[{"name":"Department of Pathology, West China Hospital, West China Medical School, Sichuan University, Chengdu, China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Linmao","family":"Zheng","sequence":"additional","affiliation":[{"name":"Department of Pathology, West China Hospital, West China Medical School, Sichuan University, Chengdu, China"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mei","family":"Kang","sequence":"additional","affiliation":[{"name":"Department of Laboratory Medicine, West China Hospital, Sichuan University, Chengdu, China;"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ting","family":"Liu","sequence":"additional","affiliation":[{"name":"Department of Hematology, West China Hospital, Sichuan University, Chengdu, China;"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Fang","family":"He","sequence":"additional","affiliation":[{"name":"Center of Infectious Diseases, West China Hospital, Sichuan University, Chengdu, China;"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"2795","reference":[{"key":"b1","series-title":"Lancet","first-page":"951","article-title":"Leishmaniasis","volume":"117","author":"Burza","year":"2018"},{"key":"b2","series-title":"Lancet","doi-asserted-by":"publisher","first-page":"312","DOI":"10.1016\/S0140-6736(16)30171-4","article-title":"Neglected tropical diseases: progress towards addressing the chronic pandemic","volume":"389","author":"Molyneux","year":"2017"},{"key":"b3","series-title":"Parasit Vectors","doi-asserted-by":"publisher","first-page":"31","DOI":"10.1186\/1756-3305-5-31","article-title":"Current epidemiological profile and features of visceral leishmaniasis in People\u2019s Republic of China","volume":"5","author":"Wang","year":"2012"},{"key":"b4","series-title":"Blood","doi-asserted-by":"publisher","first-page":"e151","DOI":"10.1182\/blood-2010-08-300046","article-title":"Gene expression profiling of peripheral blood mononuclear cells from children with active hemophagocytic lymphohistiocytosis","volume":"117","author":"Sumegi","year":"2011"},{"key":"b5","series-title":"Blood","doi-asserted-by":"publisher","first-page":"2476","DOI":"10.1182\/blood-2009-11-255174","article-title":"Sustained IL-4 exposure leads to a novel pathway for hemophagocytosis, inflammation, and tissue macrophage accumulation","volume":"116","author":"Milner","year":"2010"},{"key":"b6","series-title":"Front Immunol","first-page":"239","article-title":"Protection from inflammatory organ damage in a murine model of hemophagocytic lymphohistiocytosis using treatment with IL-18 binding protein","volume":"3","author":"Chiossone","year":"2012"},{"key":"b7","series-title":"Immunol Allergy Clin North Am","doi-asserted-by":"publisher","first-page":"293","DOI":"10.1016\/j.iac.2008.01.010","article-title":"Hemophagocytic lymphohistiocytosis and other hemophagocytic disorders","volume":"28","author":"Filipovich","year":"2008"},{"key":"b8","series-title":"Ann Hematol","doi-asserted-by":"publisher","first-page":"1143","DOI":"10.1007\/s00277-011-1367-0","article-title":"Hemophagocytic syndrome associated with visceral leishmaniasis in an immunocompetent adult-case report and review of the literature","volume":"91","author":"Koubaa","year":"2012"},{"key":"b9","series-title":"Rev Cubana Med Trop","first-page":"e524","article-title":"Hemophagocytic lymphohistiocytosis associated with visceral leishmaniasis. 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