loading page

Second malignant neoplasms following treatment for hepatoblastoma: an international report and review of the literature
  • +7
  • Angela Trobaugh-Lotrario,
  • Kenichiro Watanabe,
  • Allison O'Neill,
  • Bożenna Dembowska-Bagińska,
  • Beate Haeberle,
  • Eiso Hiyama,
  • Piotr Czauderna,
  • Rebecka Meyers,
  • Max Langham,
  • James Feusner
Angela Trobaugh-Lotrario
Sacred Heart Medical Center

Corresponding Author:angela.trobaugh@providence.org

Author Profile
Kenichiro Watanabe
Shizuoka Children's Hospital
Author Profile
Allison O'Neill
Dana-Farber/Children's Hospital Cancer Center
Author Profile
Bożenna Dembowska-Bagińska
The Children's Memorial Health Institute
Author Profile
Beate Haeberle
University of Munich
Author Profile
Eiso Hiyama
Hiroshima University
Author Profile
Piotr Czauderna
Gdanski Uniwersytet Medyczny
Author Profile
Rebecka Meyers
Primary Childrens Hospital
Author Profile
Max Langham
University of Tennessee Health Science Center College of Medicine
Author Profile
James Feusner
Childrens Hospital Oakland
Author Profile

Abstract

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.