Background Survivors of childhood cancer are at risk of subsequent primary neoplasms (SPNs), but the risk of developing specific digestive SPNs beyond age 40 years remains uncertain. We investigated risks of specific digestive SPNs within the largest available cohort worldwide.
Methods The PanCareSurFup cohort includes 69 460 five-year survivors of childhood cancer from 12 countries in Europe. Risks of digestive SPNs were quantified using standardised incidence ratios (SIRs), absolute excess risks and cumulative incidence.
Results 427 digestive SPNs (214 colorectal, 62 liver, 48 stomach, 44 pancreas, 59 other) were diagnosed in 413 survivors. Wilms tumour (WT) and Hodgkin lymphoma (HL) survivors were at greatest risk (SIR 12.1; 95% CI 9.6 to 15.1; SIR 7.3; 95% CI 5.9 to 9.0, respectively). The cumulative incidence increased the most steeply with increasing age for WT survivors, reaching 7.4% by age 55% and 9.6% by age 60 years (1.0% expected based on general population rates). Regarding colorectal SPNs, WT and HL survivors were at greatest risk; both seven times that expected. By age 55 years, 2.3% of both WT (95% CI 1.4 to 3.9) and HL (95% CI 1.6 to 3.2) survivors had developed a colorectal SPN—comparable to the risk among members of the general population with at least two first-degree relatives affected.
Conclusions Colonoscopy surveillance before age 55 is recommended in many European countries for individuals with a family history of colorectal cancer, but not for WT and HL survivors despite a comparable risk profile. Clinically, serious consideration should be given to the implementation of colonoscopy surveillance while further evaluation of its benefits, harms and cost-effectiveness in WT and HL survivors is undertaken.
- CANCER EPIDEMIOLOGY
- COLORECTAL CANCER SCREENING
- COLORECTAL CANCER
- GASTROINTESTINAL CANCER
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Deceased Deceased 27th November 2018, to whom this work is dedicated.
Contributors RCR, MH, EB, JB, SG, DG, PK, MT, FW, TW, CS, ZJ, RH, PL, LZZ, CK, JFW, FdV, LK, LH: study design and concept. RCR, KW, DW, MH: statistical analyses. RCR, MH: drafting of manuscript. RCR, KW, CB, DW, DA, RS, FB, EB, AB, JB, EAMF, MMF-B, ID, SG, DG, TG, JG, NH, SH, MJ, PK, MK, RK, HL, HØ, CR, E-MH, RS, FEvL, JT CV, ZW, GD, DI, MT, GV-B, FW, TW, CS, ZJ, RH, PL, LZZ, CK, JFW, FdV, LK, LH, MH: Interpretation of data and critically revising of manuscript. All authors read and approved the final version of the manuscript.
Funding The PanCareSurFup consortium and related work was supported by the European Union’s Seventh Framework Programme for research, technological development and demonstration under grant agreement no. 257 505. Additional financial support was received from: The Fondation Force de recherche sur le cancer de l'enfant (FORCE), The Italian Association for Cancer Research and the Compagnia San Paolo; The Fondo Chiara Rama ONLUS; The Swedish Childhood Cancer Fund; the French Association for Cancer Research (ARC); The French National Agency For Research (ANR) (Hope-Epi project); the French National Cancer Institute (INCA); Pfizer Foundation for Children and Adolescent Health; Slovenian Research Agency; the Swiss Paediatric Oncology Group; The Swiss Cancer League (KLS-3412-02-2014, KLS-3886-02-2016); The Swiss Cancer Research foundation (KFS-02783-02-2011, KFS-4157-02-2017); The Swiss National Science Foundation (PDFMP3_141775), The Dutch Cancer Society, The Norwegian Childhood Cancer Foundation, and Children with Cancer UK (grant no: 20457).
Competing interests HL and TW are shareholders in, and have signed an intellectual property agreement with the company Concidera Health. The company develops clinical decision support tools for childhood cancer survivorship.
Patient consent for publication Not required.
Ethics approval Ethical approval was not obtained specifically for this study as it involved pooling of non-identifiable data. Ethical approval was obtained within the country of origin of each contributing subcohort separately.
Provenance and peer review Not commissioned; externally peer reviewed.
Data availability statement Data are available on reasonable request. The data are not publicly available due to them containing semi-identifiable information that could compromise research participant privacy. Nonetheless, additional summary tables of count data or person-years are available from the corresponding author on request.
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