Background: Fusarium is a conditional pathogen that can cause invasive infection in patients with hematological diseases under immune function. Methods: A case of recurrent and refractory Philadelphia chromosome-positive acute lymphoblastic leukemia was treated with allogeneic hematopoietic stem cell transplantation after chimeric antigen receptor-modified T cells treatment. Results: During transplantation, disseminated Fusarium infection occurred, involving the skin, liver, spleen and central nervous system, and the patient eventually died. Conclusions: Early identification of Fusarium infection based on the characteristic rash and timely antifungal treatment can improve the cure rate.