TY - JOUR
T1 - Risk of soft-tissue sarcoma among 69 460 five-year survivors of childhood cancer in Europe
AU - Bright, Chloe J
AU - Hawkins, Mike M
AU - Winter, David L
AU - Alessi, Daniela
AU - Allodji, Rodrigue S
AU - Bagnasco, Francesca
AU - Bárdi, Edit
AU - Bautz, Andrea
AU - Byrne, Julianne
AU - Feijen, Elizabeth A M
AU - Fidler, Miranda M
AU - Garwicz, Stanislaw
AU - Grabow, Desiree
AU - Gudmundsdottir, Thorgerdur
AU - Guha, Joyeeta
AU - Haddy, Nadia
AU - Jankovic, Momcilo
AU - Kaatsch, Peter
AU - Kaiser, Melanie
AU - Kuehni, Claudia E
AU - Linge, Helena
AU - Øfstaas, Hilde
AU - Ronckers, Cecile M
AU - Skinner, Roderick
AU - Teepen, Jop C
AU - Terenziani, Monica
AU - Vu-Bezin, Giao
AU - Wesenberg, Finn
AU - Wiebe, Thomas
AU - Sacerdote, Carlotta
AU - Jakab, Zsuzsanna
AU - Haupt, Riccardo
AU - Lähteenmäki, Päivi
AU - Zaletel, Lorna Zadravec
AU - Kuonen, Rahel
AU - Winther, Jeanette F
AU - de Vathaire, Florent
AU - Kremer, Leontien C
AU - Hjorth, Lars
AU - Reulen, Raoul C
AU - PanCareSurFup Consortium
PY - 2018/6
Y1 - 2018/6
N2 - Background: Childhood cancer survivors are at risk of subsequent primary soft-tissue sarcomas (STS), but the risks of specific STS histological subtypes are unknown. We quantified the risk of STS histological subtypes after specific types of childhood cancer.Methods: We pooled data from 13 European cohorts, yielding a cohort of 69 460 five-year survivors of childhood cancer. Standardized incidence ratios (SIRs) and absolute excess risks (AERs) were calculated.Results: Overall, 301 STS developed compared with 19 expected (SIR = 15.7, 95% confidence interval [CI] = 14.0 to 17.6). The highest standardized incidence ratios were for malignant peripheral nerve sheath tumors (MPNST; SIR = 40.6, 95% CI = 29.6 to 54.3), leiomyosarcomas (SIR = 29.9, 95% CI = 23.7 to 37.2), and fibromatous neoplasms (SIR = 12.3, 95% CI = 9.3 to 16.0). SIRs for MPNST were highest following central nervous system tumors (SIR = 80.5, 95% CI = 48.4 to 125.7), Hodgkin lymphoma (SIR = 81.3, 95% CI = 35.1 to 160.1), and Wilms tumor (SIR = 76.0, 95% CI = 27.9 to 165.4). Standardized incidence ratios for leiomyosarcoma were highest following retinoblastoma (SIR = 342.9, 95% CI = 245.0 to 466.9) and Wilms tumor (SIR = 74.2, 95% CI = 37.1 to 132.8). AERs for all STS subtypes were generally low at all years from diagnosis (AER < 1 per 10 000 person-years), except for leiomyosarcoma following retinoblastoma, for which the AER reached 52.7 (95% CI = 20.0 to 85.5) per 10 000 person-years among patients who had survived at least 45 years from diagnosis of retinoblastoma.Conclusions: For the first time, we provide risk estimates of specific STS subtypes following childhood cancers and give evidence that risks of MPNSTs, leiomyosarcomas, and fibromatous neoplasms are particularly increased. While the multiplicative excess risks relative to the general population are substantial, the absolute excess risk of developing any STS subtype is low, except for leiomyosarcoma after retinoblastoma. These results are likely to be informative for both survivors and health care providers.
AB - Background: Childhood cancer survivors are at risk of subsequent primary soft-tissue sarcomas (STS), but the risks of specific STS histological subtypes are unknown. We quantified the risk of STS histological subtypes after specific types of childhood cancer.Methods: We pooled data from 13 European cohorts, yielding a cohort of 69 460 five-year survivors of childhood cancer. Standardized incidence ratios (SIRs) and absolute excess risks (AERs) were calculated.Results: Overall, 301 STS developed compared with 19 expected (SIR = 15.7, 95% confidence interval [CI] = 14.0 to 17.6). The highest standardized incidence ratios were for malignant peripheral nerve sheath tumors (MPNST; SIR = 40.6, 95% CI = 29.6 to 54.3), leiomyosarcomas (SIR = 29.9, 95% CI = 23.7 to 37.2), and fibromatous neoplasms (SIR = 12.3, 95% CI = 9.3 to 16.0). SIRs for MPNST were highest following central nervous system tumors (SIR = 80.5, 95% CI = 48.4 to 125.7), Hodgkin lymphoma (SIR = 81.3, 95% CI = 35.1 to 160.1), and Wilms tumor (SIR = 76.0, 95% CI = 27.9 to 165.4). Standardized incidence ratios for leiomyosarcoma were highest following retinoblastoma (SIR = 342.9, 95% CI = 245.0 to 466.9) and Wilms tumor (SIR = 74.2, 95% CI = 37.1 to 132.8). AERs for all STS subtypes were generally low at all years from diagnosis (AER < 1 per 10 000 person-years), except for leiomyosarcoma following retinoblastoma, for which the AER reached 52.7 (95% CI = 20.0 to 85.5) per 10 000 person-years among patients who had survived at least 45 years from diagnosis of retinoblastoma.Conclusions: For the first time, we provide risk estimates of specific STS subtypes following childhood cancers and give evidence that risks of MPNSTs, leiomyosarcomas, and fibromatous neoplasms are particularly increased. While the multiplicative excess risks relative to the general population are substantial, the absolute excess risk of developing any STS subtype is low, except for leiomyosarcoma after retinoblastoma. These results are likely to be informative for both survivors and health care providers.
U2 - 10.1093/jnci/djx235
DO - 10.1093/jnci/djx235
M3 - Article
C2 - 29165710
SN - 0027-8874
VL - 110
SP - 649
EP - 660
JO - Journal of the National Cancer Institute
JF - Journal of the National Cancer Institute
IS - 6
ER -
