Background: Treatment intensification has improved survival in patients with hepatoblastoma (HB); however, these treatments are associated with an increased risk of late effects including second malignant neoplasms (SMNs). Data is limited regarding SMNs following HB treatment. Methods: Cases of SMNs following treatment for HB reported in the literature and from personal communication were analyzed to further assess this late effect. Results: Thirty-eight patients were identified. Median age at diagnosis of HB was 16 months (range: 3 to 168 months). All patients had received a platinum agent, and almost all had anthracycline exposure. Of 12 patients with a known history of liver transplantation for primary resection of their HB, the majority had post-transplant lymphoproliferative disorder (PTLD) (n=7). The most common SMNs reported were non-PTLD hematopoietic malignancies (n=19). Solid tumors were seen in 12 patients: peripheral neuroectodermal tumor/Ewing sarcoma (3); and one each for renal cell carcinoma, nephroblastoma, colorectal carcinoma, thyroid carcinoma, medulloblastoma, clear cell sarcoma-like tumor, hepatocellular carcinoma, osteosarcoma, and malignant schwannoma. Of 36 patients with data, nineteen survived. Conclusions: SMNs following HB treatment were seen in patients with anthracycline (and cisplatin) exposure, hereditary tumor predisposition syndromes, and/or history of liver transplantation. Hematopoietic malignancies were the most common SMN reported in this cohort and were diagnosed earlier than other SMNs. Prospective collection of data via a companion late effects study or international registry could be used to further evaluate rates and risks of SMNs as well as tumor predisposition syndromes in patients treated for HB.