Background: Hepatosplenic fungal infection in patients with acute leukemia is a rare disease. Case Report: We report a 33-year-old female with acute myeloid leukemia (AML) who developed probable hepatosplenic fungal infection after induction chemotherapy, underwent hematopoietic stem cell transplantation (HSCT) from matched sibling brother successfully. The patient received anti-fungal treatment for seven months and lesions tended to stabilize before transplantation. She was also treated with voriconazole and caspofungin throughout transplantation. Repeat abdominal MRI post-transplant revealed obvious regression of hepatic and splenic lesions. Eventually, she was discharged with full recovery of cell count. Conclusion: Complete imaging resolution of lesions is not a prerequisite for HSCT if the infection can be controlled with antifungal therapy.