The case was a 15-year-old male patient from Linzhou, Henan province and he was a student. Due to “intermittent fever for more than half a month”, he went to a local municipal hospital on March 3, 2021. The blood routine test showed leukocyte was 2.60 × 10⁹/L, platelet was 65 × 10⁹/L and hemoglobin was 113 g/L. He was diagnosed with infectious fever with leukothrombopenia and splenomegaly. After anti-infection treatment, the symptoms were improved and he was discharged home. But the condition sometimes recurred and he was admitted to the hematology department of a provincial hospital because of “repeated fever with fatigue and pancytopenia for more than 1 year” on April 19, 2022. The blood routine test showed white blood cell was 1.79 × 10⁹/L, hemoglobin was 73 g/L and platelet was 54 × 10⁹/L. Ultrasound showed enlarged lymphnodes in the neck and axilla, large liver and spleen with widened splenic veins. Bone marrow smear showed hyperplasia was active, granulocyte series reduced, erythrocyte series was significantly active while mature erythrocytes clustered and arranged in row, and no megakaryocyte was found in this smear. Bone marrow biopsy showed extensive fibrosis, increasing proportion of erythrocyte series, many megakaryocyte cells and also showed T cells were easy to be seen with scattered individual large cells. Positron emission tomography-computed tomography (PET-CT) showed diffuse active in splenic metabolism and no abnormalities in hepatic metabolism. Then the patient was suspected to have a splenic lymphoma and was transferred to surgery for splenectomy + abdominal lymph node dissection. Pathology of the spleen showed moderate histiocytic hyperplasia and pathogen in the cytoplasm of the histocyte which was verified Leishmania donovani by gene sequencing. Pathology of the spleen also showed extramedullary hematopoietic. The patient was diagnosed with visceral leishmaniasis. The patient had a history of contact with dogs and the county where he lived had visceral leishmaniasis case report. He was admitted to the Fifth People’s Hospital of Anyang in June 27, 2022 and was began to be treated with sodium stibogluconate injection from that time. The admission examination showed emaciation, malnutrition and hepatomegaly that liver could be palpated 10cm below costal margin on right midclavicular line, moderate quality, no tenderness. Blood routine test showed that the white blood cell was 11.17 × 10⁹/L， hemogloubin was 10⁴ g/L and platelet was 245 × 10⁹/L. The result of rK39 kala-azar detect rapid test was positive. Abdominal CT showed a significantly enlarged liver. After treated with sodium stibogluconate injection for 1 course (0.6 g/d, 9 d), The temperature returned to normal and the general condition improved, but Leishmania amastigote was visible in his bone marrow smear. So another course sodium stibogluconate injection (0.6 g/d, 9 d) was given to him. After the second course’s treatment, the bone marrow smear was reexamined and no Leishmania amastigote could be found. The paitent was recovered and discharged home. He felt well and the abdominal CT showed liver was somewhat reduced and bone marrow trephine biopsy performed had returned to normal and bone marrow smear was not found Leishmania amastigote about 10 months after initiation of the therapy.