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Peer-reviewedCC BYJNCI: Journal of the National Cancer Institute

Risk of subsequent primary melanoma in 68 002 five-year survivors of childhood and adolescent cancer in Europe: the PanCareSurFup cohort study

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Abstract

BACKGROUND: Survivors of childhood cancer are at risk of subsequent primary melanoma, but the magnitude of the risk after different childhood cancer types and beyond age 50 years remains inadequately characterized. We quantified risks in the largest cohort of childhood cancer survivors worldwide. METHODS: The PanCare Childhood and Adolescent Cancer Survivor Care and Follow-Up Studies cohort includes 68 002 five-year childhood cancer survivors across Europe (diagnosed 1940-2008). Subsequent primary melanoma risks were quantified using standardized incidence ratios (SIRs), absolute excess risks (AERs), cumulative incidence, and relative risks (RRs). RESULTS: Over 1 239 675 person-years, 197 subsequent primary melanoma were ascertained, whereas 84.9 were expected (SIR = 2.3, 95% confidence interval [CI] = 2.0 to 2.7). Although the standardized incidence ratio decreased with attained age (P for trend < .001), it remained increased at 2.1-fold (95% CI = 1.5 to 3.1) at age 50 years and older. By age 65 years, cumulative incidence was 1.2% (0.8% expected). Of all cancer types, heritable retinoblastoma survivors had the greatest risk (SIR = 16.5, 95% CI = 10.8 to 25.3; AER = 94.4, 95% CI = 59.9 to 148.9), with cumulative incidence of 3.3% at age 50 years and 5.5% at age 60 years. Survivors treated with radiotherapy had a 70% greater risk than those treated without (RR = 1.7, 95% CI = 1.1 to 2.7), increasing to 4.0-fold by age 50 years and older (RR = 3.9, 95% CI = 1.1 to 13.9). For chemotherapy, no statistically significant association was found (RR = 0.9, 95% CI = 0.5 to 1.6). CONCLUSIONS: Childhood cancer survivors, particularly those with heritable retinoblastoma and those treated with radiotherapy, remain at increased risk of melanoma beyond age 50 years. Current survivorship guidelines focus principally on treatment history; however, our findings suggest that long-term follow-up guidelines should additionally include childhood cancer type and attained age as risk-stratifying factors, particularly where individual treatment records are unavailable.

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BibTeX
@article{shojaie2026risk,
  title = {{Risk of subsequent primary melanoma in 68 002 five-year survivors of childhood and adolescent cancer in Europe: the PanCareSurFup cohort study}},
  author = {Behnoush Shojaie and Jop C Teepen and David L Winter and Mike P Gardner and Melanie Kaiser and Desiree Grabow and Cécile M Ronckers and Julianne Byrne and Roderick Skinner and Daniela Alessi and Hama Baqaeen and Rodrigue S Allodji and Helena J H van der Pal and Momcilo Jankovic and Francesca Bagnasco and Eva Steliarova-Foucher and Ben D Spycher and Grit Sommer and Edit Bárdi and Thorgerdur Gudmundsdottir and Milena M Maule and Gisela Michel and Monica Muraca and Maria W Gunnes and Line Kenborg and Henrik Hjalgrim and Carlotta Sacerdote and Zsuzsanna Jakab and Riccardo Haupt and Thomas Wiebe and Monica Terenziani and Lorna Zadravec Zaletel and Leontien C M Kremer and Claudia E Kuehni and Päivi M Lähteenmäki and Florent de Vathaire and Lars Hjorth and Michael M Hawkins and Raoul C Reulen},
  journal = {JNCI: Journal of the National Cancer Institute},
  year = {2026},
  month = nov,
  doi = {10.1093/jnci/djag094},
  url = {https://hal.science/hal-05851609v1},
  note = {CC BY},
}
RIS
TY  - JOUR
TI  - Risk of subsequent primary melanoma in 68 002 five-year survivors of childhood and adolescent cancer in Europe: the PanCareSurFup cohort study
AU  - Behnoush Shojaie
AU  - Jop C Teepen
AU  - David L Winter
AU  - Mike P Gardner
AU  - Melanie Kaiser
AU  - Desiree Grabow
AU  - Cécile M Ronckers
AU  - Julianne Byrne
AU  - Roderick Skinner
AU  - Daniela Alessi
AU  - Hama Baqaeen
AU  - Rodrigue S Allodji
AU  - Helena J H van der Pal
AU  - Momcilo Jankovic
AU  - Francesca Bagnasco
AU  - Eva Steliarova-Foucher
AU  - Ben D Spycher
AU  - Grit Sommer
AU  - Edit Bárdi
AU  - Thorgerdur Gudmundsdottir
AU  - Milena M Maule
AU  - Gisela Michel
AU  - Monica Muraca
AU  - Maria W Gunnes
AU  - Line Kenborg
AU  - Henrik Hjalgrim
AU  - Carlotta Sacerdote
AU  - Zsuzsanna Jakab
AU  - Riccardo Haupt
AU  - Thomas Wiebe
AU  - Monica Terenziani
AU  - Lorna Zadravec Zaletel
AU  - Leontien C M Kremer
AU  - Claudia E Kuehni
AU  - Päivi M Lähteenmäki
AU  - Florent de Vathaire
AU  - Lars Hjorth
AU  - Michael M Hawkins
AU  - Raoul C Reulen
PY  - 2026
DA  - 2026/11/04
JO  - JNCI: Journal of the National Cancer Institute
DO  - 10.1093/jnci/djag094
UR  - https://hal.science/hal-05851609v1
AB  - BACKGROUND: Survivors of childhood cancer are at risk of subsequent primary melanoma, but the magnitude of the risk after different childhood cancer types and beyond age 50 years remains inadequately characterized. We quantified risks in the largest cohort of childhood cancer survivors worldwide. METHODS: The PanCare Childhood and Adolescent Cancer Survivor Care and Follow-Up Studies cohort includes 68 002 five-year childhood cancer survivors across Europe (diagnosed 1940-2008). Subsequent primary melanoma risks were quantified using standardized incidence ratios (SIRs), absolute excess risks (AERs), cumulative incidence, and relative risks (RRs). RESULTS: Over 1 239 675 person-years, 197 subsequent primary melanoma were ascertained, whereas 84.9 were expected (SIR = 2.3, 95% confidence interval [CI] = 2.0 to 2.7). Although the standardized incidence ratio decreased with attained age (P for trend < .001), it remained increased at 2.1-fold (95% CI = 1.5 to 3.1) at age 50 years and older. By age 65 years, cumulative incidence was 1.2% (0.8% expected). Of all cancer types, heritable retinoblastoma survivors had the greatest risk (SIR = 16.5, 95% CI = 10.8 to 25.3; AER = 94.4, 95% CI = 59.9 to 148.9), with cumulative incidence of 3.3% at age 50 years and 5.5% at age 60 years. Survivors treated with radiotherapy had a 70% greater risk than those treated without (RR = 1.7, 95% CI = 1.1 to 2.7), increasing to 4.0-fold by age 50 years and older (RR = 3.9, 95% CI = 1.1 to 13.9). For chemotherapy, no statistically significant association was found (RR = 0.9, 95% CI = 0.5 to 1.6). CONCLUSIONS: Childhood cancer survivors, particularly those with heritable retinoblastoma and those treated with radiotherapy, remain at increased risk of melanoma beyond age 50 years. Current survivorship guidelines focus principally on treatment history; however, our findings suggest that long-term follow-up guidelines should additionally include childhood cancer type and attained age as risk-stratifying factors, particularly where individual treatment records are unavailable.
SN  - 0027-8874
SN  - 1460-2105
ER  -