A case of pediatric visceral leishmaniasis-related hemophagocytic lymphohistiocytosis diagnosed by mNGS. (August 2020)

• Record Type: Journal Article
• Authors: Guo, Fang; Kang, Lei; Xu, Meixian
• Is Part Of: International journal of infectious diseases. Volume 97(2020)
• Page Start: 27
• Page End: 29
• Publication Date: 2020-08
• Subjects: VL visceral leishmaniasis -- HLH hemophagocytic lymphohistiocytosis -- VL-HLH visceral leishmaniasis-related hemophagocytic lymphohistiocytosis -- mNGS next-generation meta-genome sequencing -- BMA bone marrow aspiration -- AMB libosomal amphotericin B; Visceral leishmaniasis-related hemophagocytic lymphohistiocytosis -- Next-generation meta-genome sequencing -- Bone marrow aspiration; Communicable diseases -- Periodicals; Communicable Diseases -- Periodicals; Communicable diseases; Periodicals; Electronic journals; 616.9
• DOI: 10.1016/j.ijid.2020.05.056
• Languages: English
• ISSNs: 1201-9712

Abstract

Highlights: Atypical clinical features make it hard to identify VL in HLH in non-epidemic area. VL is difficult to diagnose when traditional tests are ineffective, while mNGS is. mNGS can be used as a detection method of HLH in children caused by rare pathogens. Abstract: Background: Visceral leishmaniasis-related hemophagocytic lymphohistiocytosis (VL-HLH) is a secondary hemophagocytic syndrome, which can be life-threatening, caused by leishmania and transmitted by infected sandflies. Rapid and accurate identification of leishmania is crucial for clinical strategies. Case report: Here, we report an infantile infection in a non-epidemic area of China. The infant was a 9.5-month-old girl with fever, pancytopenia and hepatosplenomegaly, which meet the HLH-2004 standard, and the negative gene results exclude congenital HLH. However, chemotherapy is ineffective and is accompanied by severe infection. Fortunately, she is diagnosed with VL-HLH (visceral leishmaniasis-related hemophagocytic lymphohistiocytosis), as leishmania is detected by next-generation meta-genome sequencing (mNGS) and quickly relieved after treatment with libosomal amphotericin B (L-AMB). Conclusion: mNGS can detect leishmania in pediatric HLH, and should be performed as a new detection for VL-HLH, particularly for infants, who may not respond to HLH-2004 regimen.