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Post-transplant lymphoproliferative disorder (PTLD) is the most common form of lymphoproliferation in childhood and is associated with significant morbidity and mortality. In this report we reviewed the case of a pediatric patient who experienced PTLD after allogeneic hematopoietic stem cell transplantation (HSCT) from a human leukocyte antigen (HLA)-identical sibling.The clinical characteristics, diagnosis, and treatment of PTLD after sibling HSCT in a 4-year-old boy with severe β-thalassemia was retrospectively reviewed.Medical records revealed the patient developed a fever and superficial lymphadenopathy and soft palate enlargement 8 months post-HSCT. Pathologic diagnosis indicated non-Hodgkin lymphoma (B-cell type), which resulted in a reduced dose of immunosuppressant and the initiation of chemotherapy (administered according to the BFM95 protocol for 2 courses; 4 courses of rituximab therapy was also administered). Currently, the patient has been disease-free for over 3 years. There are no specific guidelines for the treatment of PTLD. The status of stem cell implantation after transplantation, and graft versus host disease should be evaluated jointly, and rituximab therapy and chemotherapy with BFM-95 may be used for treatment of pediatric PTLD after HSCT.The current case represents a unique opportunity to review a pediatric patient with β-thalassemia. The successful treatment of post-transplant non-Hodgkin B lymphoma may help other physicians in the management of similar pediatric cases.