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We aimed to describe the clinical and laboratory findings of HLH patients secondary to Visceral leishmaniasis (VL) and their treatment outcome during a 4-year follow-up period compared to primary HLH.<\/jats:p><\/jats:sec><jats:sec><jats:title>Method<\/jats:title><jats:p>Forty children with primary HLH confirmed by genetic study and 20 children with HLH secondary to VL confirmed by a blood or bone marrow polymerase chain reaction from 2014 to 2018 in Shiraz, Fars province, Southern Iran, were enrolled.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>The median age at diagnosis was 11.5\u2009months (range 1\u2013170), and 56.7% were male. Fever and splenomegaly were the most frequent clinical presentations. 93.3% of the subjects had an HScore &gt;\u2009169, which had a good correlation with HLH-2004 criteria (<jats:italic>r<\/jats:italic>\u2009=\u20090.371,<jats:italic>P<\/jats:italic>\u2009=\u20090.004). Patients with primary HLH experienced more thrombocytopenia (<jats:italic>P<\/jats:italic>\u2009=\u20090.012) and higher alanine transaminase (<jats:italic>P<\/jats:italic>\u2009=\u20090.016), while patients with VL-associated HLH had higher ferritin (<jats:italic>P<\/jats:italic>\u2009=\u20090.034) and erythrocyte sedimentation rate (<jats:italic>P<\/jats:italic>\u2009=\u20090.011). Central nervous system (CNS) involvement occurred in 38.3% of patients. The mortality rate was higher in patients with CNS disease (61% vs. 35%,<jats:italic>P<\/jats:italic>\u2009=\u20090.051). The 3-yr overall survival rate was 35.9%. (24% in primary HLH and 100% in VL-associated HLH,<jats:italic>P<\/jats:italic>\u2009&lt;\u20090.001). In Cox regression analysis, platelet count &lt;\u2009100,000\/<jats:italic>\u03bc<\/jats:italic>l (hazard ratio 4.472, 95% confidence interval 1.324\u201315.107,<jats:italic>P<\/jats:italic>\u2009=\u20090.016) correlated with increased mortality in patients with primary HLH.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusion<\/jats:title><jats:p>VL is a potential source of secondary HLH in regions with high endemicity. Treatment of the underlying disease in VL-associated HLH is sufficient in most cases, with no need to start etoposide-based chemotherapy.<\/jats:p><\/jats:sec>","DOI":"10.1186\/s12879-021-06408-w","type":"journal-article","created":{"date-parts":[[2021,8,2]],"date-time":"2021-08-02T15:03:56Z","timestamp":1627916636000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":28,"title":["Comparison of the clinical features and outcome of children with hemophagocytic lymphohistiocytosis (HLH) secondary to visceral leishmaniasis and primary HLH: a single-center study"],"prefix":"10.1186","volume":"21","author":[{"given":"Hadi","family":"Mottaghipisheh","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kurosh","family":"Kalantar","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ali","family":"Amanati","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mansoureh","family":"Shokripour","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mahdi","family":"Shahriari","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Omid Reza","family":"Zekavat","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Soheila","family":"Zareifar","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mehran","family":"Karimi","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sezaneh","family":"Haghpanah","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mohammadreza","family":"Bordbar","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2021,8,2]]},"reference":[{"issue":"1 Suppl","key":"6408_CR1","doi-asserted-by":"publisher","first-page":"S82","DOI":"10.1016\/j.bbmt.2009.11.014","volume":"16","author":"A Filipovich","year":"2010","unstructured":"Filipovich A, McClain K, Grom A. 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Also, all methods were carried out in accordance with Declaration of Helsinki.A written informed consent form was signed by the parents or the legal guardians of the participants.","order":2,"name":"Ethics","group":{"name":"EthicsHeading","label":"Ethics approval and consent to participate"}},{"value":"Not applicable.","order":3,"name":"Ethics","group":{"name":"EthicsHeading","label":"Consent for publication"}},{"value":"The authors declare that they have no conflict of interest.","order":4,"name":"Ethics","group":{"name":"EthicsHeading","label":"Competing interests"}}],"article-number":"732"}}