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Risk of Soft-Tissue Sarcoma Among 69 460 Five-Year Survivors of Childhood Cancer in Europe

Bright, Chloe J; Hawkins, Mike M; Winter, David L; Alessi, Daniela; Allodji, Rodrigue S; Bagnasco, Francesca; Bárdi, Edit; Bautz, Andrea; Byrne, Julianne and Feijen, Elizabeth A M, et al. (2018) In Journal of the National Cancer Institute 110(6).
Abstract

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),... (More)

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.

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Journal of the National Cancer Institute
volume
110
issue
6
pages
12 pages
publisher
Oxford University Press
external identifiers
  • scopus:85054233382
ISSN
1460-2105
DOI
10.1093/jnci/djx235
language
English
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yes
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ffaaca2a-fb8a-4e3e-b9d9-1f7fd6ca922a
date added to LUP
2018-05-08 14:35:06
date last changed
2019-06-19 03:54:50
@article{ffaaca2a-fb8a-4e3e-b9d9-1f7fd6ca922a,
  abstract     = {<p>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.</p><p>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.</p><p>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 &lt; 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.</p><p>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.</p>},
  articleno    = {djx235},
  author       = {Bright, Chloe J and Hawkins, Mike M and Winter, David L and Alessi, Daniela and Allodji, Rodrigue S and Bagnasco, Francesca and Bárdi, Edit and Bautz, Andrea and Byrne, Julianne and Feijen, Elizabeth A M and Fidler, Miranda M and Garwicz, Stanislaw and Grabow, Desiree and Gudmundsdottir, Thorgerdur and Guha, Joyeeta and Haddy, Nadia and Jankovic, Momcilo and Kaatsch, Peter and Kaiser, Melanie and Kuehni, Claudia E and Linge, Helena and Øfstaas, Hilde and Ronckers, Cecile M and Skinner, Roderick and Teepen, Jop C and Terenziani, Monica and Vu-Bezin, Giao and Wesenberg, Finn and Wiebe, Thomas and Sacerdote, Carlotta and Jakab, Zsuzsanna and Haupt, Riccardo and Lähteenmäki, Päivi and Zaletel, Lorna Zadravec and Kuonen, Rahel and Winther, Jeanette F and de Vathaire, Florent and Kremer, Leontien C and Hjorth, Lars and Reulen, Raoul C and , },
  issn         = {1460-2105},
  language     = {eng},
  number       = {6},
  pages        = {12},
  publisher    = {Oxford University Press},
  series       = {Journal of the National Cancer Institute},
  title        = {Risk of Soft-Tissue Sarcoma Among 69 460 Five-Year Survivors of Childhood Cancer in Europe},
  url          = {http://dx.doi.org/10.1093/jnci/djx235},
  volume       = {110},
  year         = {2018},
}