A 31-year-old male patient hailing from Xianfeng County, Hubei Province, was presented. He had gone to work in Yangquan, Shanxi Province on July 2022 and returned to Xianfeng on January 2023. The patient developed a fever on February, and received intravenous therapy at a local health center. On April 28, he had a relapse of fever with the highest body temperature of 39.8 ℃. Despite consultations at multiple hospitals, no definite diagnosis was made. On May 6, he was transferred to the Department of Infectious Diseases, Wuhan Jinyintan Hospital. Abdominal ultrasound scans displayed splenomegaly, and microscopic examination of bone marrow smears identified amastigotes, while rk39 immunochromatographic strip test detected positive serum antibodies against Leishmania. Genomic DNA was extracted from blood samples, and PCR assay of internal transcribed spacer 1 (ITS-1) of Leishmania ribosomal DNA amplified a band with approximately 310 bp in length. Sequencing showed a 99% homology between the amplified sequence and the corresponding sequence of L. donovani. The case was definitively diagnosed with visceral leishmaniasis, and given two cycles of treatment with sodium stibogluconate injection (600 mg/d for 6 d each cycle, with a one-week interval between cycles) since May 8. The patients had goo recovery at the three-month follow-up visit post-treatment, with normal routine blood tests and no Leishmania detected on bone marrow smears.