To investigate the potential benefits of metagenomics next-generation sequencing (mNGS) on diagnosis for patients with acquired immune deficiency syndrome (AIDS) and Leishmaniasis, and to improve the diagnostic and therapeutic understanding of leishmaniasis.

The clinical records, diagnosis and treatment of a patient with AIDS complicated by hemophagocytic syndrome due to visceral leishmaniasis (VL) admitted to Zhongnan Hospital of Wuhan University on December 28th, 2021 were reviewed, retrospectively.

The patient was admitted to hospital presenting with fever. After admission, the laboratory investigations revealed peripheral blood pancytopenia, hepatorenal dysfunction, hypofibrinogenemia and markedly elevated inflammatory markers. Hemophagocytic syndrome was confirmed by bone marrow biopsy. However, no positive pathogens were found after various etiological examinations, and antibiotic therapy was ineffective. Total of 1 262 sequences of Leishmania parasites (80 sequences of Leishmania donovani and 20 sequences of Leishmania infantum) were identified from the peripheral blood of the patient through mNGS. Ultimately, a diagnosis of AIDS with concurrent visceral leishmaniasis and hemophagocytic syndrome was established. After sodium antimony gluconate (0.6 g/d, intravenous injection during a course of 10 d) and buctillavir/emtricitabine/propofol tenofovir (50 mg/200 mg/25 mg, oral) for anti-HIV treatment, the patient were improved and discharged, and no recurrence was observed during the follow-up.

mNGS is proved to be an effective diagnostic method, especially for patients with HIV/AIDS who cannot be conclusively diagnosed through conventional laboratory tests. HAART at an early stage of AIDS patients is of significant importance for improving the prognosis and preventing the recurrence of VL.