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Diagnosis and Management of Visceral Leishmaniasis in Children: A French Retrospective Study
Justine de Larminat1, Naïma Dahane2, Nicolas Argy3,4
1General Pediatrics, Infectious Diseases and Internal Medicine, Robert Debré Hospital, Assistance Publique-Hôpitaux de Paris, Paris, France.
Insights
Pediatric visceral leishmaniasis (VL) in France is rare, often presenting with delayed diagnosis and hemophagocytic lymphohistiocytosis (HLH). Combined blood and bone marrow PCR significantly improves diagnosis, with liposomal amphotericin B proving effective for treatment.
Area of Science:
- Pediatric infectious diseases
- Parasitology
- Clinical diagnostics
Background:
- Pediatric visceral leishmaniasis (VL) diagnosis and management in high-income countries are under-researched.
- This study focuses on pediatric VL cases in mainland France.
Purpose of the Study:
- To investigate the characteristics, diagnostic approaches, and treatment outcomes of pediatric VL in France.
- To evaluate the effectiveness of diagnostic tools and therapeutic regimens for pediatric VL.
Main Methods:
- Retrospective study of children hospitalized for VL in French university hospitals (January 2012 - August 2022).
- Data collected included clinical presentation, biological findings, diagnostic methods (PCR, smears), and treatment regimens.
- Analysis of diagnostic delays, co-morbidities like hemophagocytic lymphohistiocytosis (HLH), and treatment responses.
Main Results:
- Sixty-three pediatric VL patients were identified, with a median age of 2 years; 36 had autochthonous VL.
- Common symptoms included fever (92%) and splenomegaly (78%); 44% presented with HLH.
- Combined blood and bone marrow PCR achieved 100% positivity for VL diagnosis.
- Liposomal amphotericin B was used for treatment, with a 6.3% relapse rate and full recovery in all patients.
Conclusions:
- Pediatric VL in France is infrequent but associated with significant diagnostic delays and a high incidence of HLH.
- Combined PCR methods offer optimal diagnostic accuracy for pediatric VL.
- Standardized treatment protocols and follow-up strategies are essential for managing pediatric VL.
Background:
Few studies have investigated the diagnosis and management of pediatric visceral leishmaniasis (VL) in high-income countries.
Methods:
All children hospitalized for VL in university hospitals across mainland France between January 2012 and August 2022 were included in this retrospective study.
Results:
Sixty-three patients were included with an M/F sex ratio of 1.25 and a median age of 2 years (IQR: 1; 5.4). At least 36 patients had autochthonous VL. The median delay between symptom onset and diagnosis was 20 days (IQR: 11.7; 35.2) [1-193]. The most common symptoms were fever (92%) and splenomegaly (78%). Biological findings included inflammatory syndrome (90%), pancytopenia (79%), and hepatic cytolysis (52%). Twenty-four children (44%) presented hemophagocytic lymphohistiocytosis (HLH). Bone marrow smears, bone marrow real-time PCR (qPCR), and blood qPCR were positive for VL in 64%, 90%, and 96% of the tested patients, respectively. Positivity reached 100% in patients who underwent combined blood and bone marrow PCR. All the patients were treated with liposomal amphotericin B (chemical name, (1R,3S,5R,6R,9R,11R,15S,16R,17R,18S,19E,21E,23E,25E,27E,29E,31E,33R,35S,36R,37S)-33-[(2R,3S,4S,5S,6R)-4-amino-3,5-dihydroxy-6-methyloxan-2-yl]oxy-1,3,5,6,9,11,17,37-octahydroxy-15,16,18-trimethyl-13-oxo-14,39-dioxabicyclo[33.3.1]nonatriaconta-19,21,23,25,27,29,31-heptaene-36-carboxylic acid;2-aminoguanidine). Ten different regimens were used, ranging from 18 to 30 mg/kg cumulative doses given from 2 to 8 days. The median follow-up duration was 2.8 months. Four patients (6.3%) relapsed, one of whom was not retreated. All the children recovered.
Conclusions:
Pediatric VL remains rare in France, with long diagnostic delays and a high frequency of HLH. The combination of blood and bone marrow PCR optimizes the diagnosis. Standardization of therapeutic regimens and follow-up is necessary.
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