Background Although anthracycline therapy is invaluable for treating neoplastic disorders, morbidity includes severe cardiomyopathy that leads to heart transplantation. This multicenter study describes the course of children who experienced anthracycline cardiomyopathy (ACM) and who subsequently required heart transplantation. Methods We reviewed transplant databases/registries at 4 pediatric heart transplant centers to identify children with ACM who were listed for heart transplantation. We reviewed medical records to determine cancer therapy, clinical course, and outcome. Results Eighteen patients were listed, and 17 underwent transplantation. Mean age at cancer diagnosis was 6.0 years (SD, 3.7). The mean anthracycline dose was 361 mg/m2 (SD, 110). The median time from cancer diagnosis to listing for heart transplantation was 9.2 years (range, 0.4-15.2 years). Six transplantations were performed in patients who had disease-free intervals of <5 years. Two patients were lost to follow-up, and 8 are alive at 4.9 years (SD, 2.0; range, 1.3-7.4 years) after transplantation. Seven patients died at 4.7 years (SD, 2.0; range, 1.2-7.1 years) after transplantation. One patient had recurrent cancer. One-, 2- and 5-year survivals were 100%, 92%, and 60%, respectively. Conclusions Cardiomyopathy that progresses to the need for heart transplantation occurs in patients receiving a wide range of cumulative anthracycline doses. The time from chemotherapy to ACM varies. Outcomes after transplantation are acceptable, and cancer recurrence is rare. Reconsideration of the 5-year disease-free wait period is warranted.