Cases of secondary acute myeloid leukemia (AML) occurring after treatment for an Ewing's sarcoma are uncommon. Therapy-related AML with t(8;21) translocation is an entity which has been well characterized. A case of AML-2 with t(8;21) and t(3;15) occurring 4 years after treatment for an Ewing's sarcoma with cyclophosphamide, doxorubicin, vincristine, dactinomycin, and radiotherapy, is reported. Autologous bone marrow transplantation was performed during second remission, 23 months after diagnosis. Reverse transcriptase polymerase chain reaction of the AML1/ETO fusion gene product was performed in order to monitor the quality of the remission. The patient currently remains in remission 24 months after the bone marrow transplantation.