Observational studyBMC infectious diseases2021
Comparison of the clinical features and outcome of children with hemophagocytic lymphohistiocytosis (HLH) secondary to visceral leishmaniasis and primary HLH: a single-center study.
Observational study in BMC infectious diseases, 2021. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 18 papers.
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Who cites it
18 citing papers in PubMed, 29 citations in OpenAlex.
- Rare disease mimicking multisystem inflammatory syndrome in children.BMC pediatrics · 2025Article
- When Hepatitis Masks Visceral Leishmaniasis: A Case of Secondary HLH in a Low-Endemic Region.Clinical case reports · 2025Article
- Polymerase Chain Reaction-Based Diagnosis of Leishmaniasis Targeting Kinetoplast Minicircle DNA in Infants with Hemophagocytic Lymphohistiocytosis.The American journal of tropical medicine and hygiene · 2025Article
- Clinical and Laboratory Features of Hemophagocytic Lymphohistiocytosis in People With Disseminated Histoplasmosis.Open forum infectious diseases · 2025Article
- Promising Targeted Therapies for Hemophagocytic Lymphohistiocytosis: A Translational Perspective Based on Immunopathology.Clinical reviews in allergy & immunology · 2025Review
- Visceral leishmaniasis related secondary haemophagocytic syndrome: A case report.The Journal of international medical research · 2025Article
- Hemophagocytic lymphohistiocytosis secondary to visceral leishmaniasis in children: case report and systematic review.Frontiers in pediatrics · 2025Article
- A 5-Month-Old Infant With a Complicated Holiday Souvenir.Clinical infectious diseases : an official publication of the Infectious Diseases Society of America · 2024Article
- Article
- Association of a decreased platelet count with poor survival in patients with adult secondary hemophagocytic lymphohistiocytosis.Annals of hematology · 2024Article
- Immune response to viscerotropicFrontiers in immunology · 2024Review
- Parasitic and Fungal Triggers of Cytokine Storm Syndrome.Advances in experimental medicine and biology · 2024Review
- Article
- Secondary hemophagocytic lymphohistiocytosis in children (Review).Experimental and therapeutic medicine · 2023Review
- Case Report: Diagnosis and Treatment of Two Clinical Cases of Visceral Leishmaniasis-Related Hemophagocytic Lymphohistiocytosis.The American journal of tropical medicine and hygiene · 2023Article
- From Infection to Death: An Overview of the Pathogenesis of Visceral Leishmaniasis.Pathogens (Basel, Switzerland) · 2023Review
- Ferritin in pediatric critical illness: a scoping review.Clinical and experimental pediatrics · 2023Article
- Reactive Hemophagocytic Lymphohistiocytosis Secondary to Ovarian Adenocarcinoma: A Rare Case Report.Journal of inflammation research · 2022Article
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Authors and funding
10 authors at 1 institution in 1 country.
Funding
No grant is acknowledged in the PubMed record.
Abstract
backgroundHemophagocytic lymphohistiocytosis (HLH) is a syndrome of excessive inflammation. 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.
methodForty 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.
resultsThe median age at diagnosis was 11.5 months (range 1-170), and 56.7% were male. Fever and splenomegaly were the most frequent clinical presentations. 93.3% of the subjects had an HScore > 169, which had a good correlation with HLH-2004 criteria (r = 0.371, P = 0.004). Patients with primary HLH experienced more thrombocytopenia (P = 0.012) and higher alanine transaminase (P = 0.016), while patients with VL-associated HLH had higher ferritin (P = 0.034) and erythrocyte sedimentation rate (P = 0.011). Central nervous system (CNS) involvement occurred in 38.3% of patients. The mortality rate was higher in patients with CNS disease (61% vs. 35%, P = 0.051). The 3-yr overall survival rate was 35.9%. (24% in primary HLH and 100% in VL-associated HLH, P < 0.001). In Cox regression analysis, platelet count < 100,000/ μ l (hazard ratio 4.472, 95% confidence interval 1.324-15.107, P = 0.016) correlated with increased mortality in patients with primary HLH.
conclusionVL 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.
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