Risk of subsequent primary leukaemias among 69,460 five-year survivors of childhood cancer diagnosed from 1940 to 2008 in Europe: A cohort study within PanCareSurFup

Rodrigue S. Allodji, Mike M. Hawkins, Chloe J. Bright, Miranda M. Fidler-Benaoudia, David L. Winter, Daniela Alessi, Brice Fresneau, Neige Journy, Vera Morsellino, Edit Bárdi, Andrea Bautz, Julianne Byrne, Elizabeth (Lieke) AM Feijen, Jop C. Teepen, Giao Vu-Bezin, Carole Rubino, Stanislaw Garwicz, Desiree Grabow, Thorgerdur Gudmundsdottir, Joyeeta GuhaEva Maria Hau, Momcilo Jankovic, Peter Kaatsch, Melanie Kaiser, Helena Linge, Monica Muraca, Damien Llanas, Cristina Veres, Hilde Øfstaas, Ibrahima Diallo, Imene Mansouri, Cecile M. Ronckers, Roderick Skinner, Monica Terenziani, Finn Wesenberg, Thomas Wiebe, Carlotta Sacerdote, Zsuzsanna Jakab, Riccardo Haupt, Päivi Lähteenmäki, Lorna Zadravec Zaletel, Claudia E. Kuehni, Jeanette F. Winther, Gisela Michel, Leontien C.M. Kremer, Lars Hjorth, Nadia Haddy, Florent de Vathaire, Raoul C. Reulen

Research output: Contribution to journalArticle

Abstract

Background: Survivors of childhood cancers are at risk of developing subsequent primary leukaemias (SPLs), but the long-term risks beyond 20 years of treatment are still unclear. We investigated the risk of SPLs in five-year childhood cancer survivors using a large-scale pan-European (PanCareSurFup) cohort and evaluated variations in the risk by cancer and demographic factors. Methods: This largest-ever assembled cohort comprises 69,460 five-year childhood cancer survivors from 12 European countries. Standardised incidence ratios (SIRs) and absolute excess risks (AERs) were calculated. Results: One hundred fifteen survivors developed an SPL including 86 myeloid leukaemias (subsequent primary myeloid leukaemias [SPMLs]), 17 lymphoid leukaemias and 12 other types of leukaemias; of these SPLs, 31 (27%) occurred beyond 20 years from the first childhood cancer diagnosis. Compared with the general population, childhood cancer survivors had a fourfold increased risk (SIR = 3.7, 95% confidence interval [CI]: 3.1 to 4.5) of developing leukaemia, and eight leukaemias per 100,000 person-years (AER = 7.5, 95% CI: 6.0 to 9.2) occurred in excess of that expected. The risks remained significantly elevated beyond 20 years from the first primary malignancy (SIR = 2.4, 95% CI: 1.6 to 3.4). Overall, the risk ratio for SPML (SIR = 5.8, 95% CI: 4.6 to 7.1) was higher than that for other SPLs. Conclusions: We demonstrate that beyond 20 years after childhood cancer diagnosis, survivors experience an increased risk for SPLs compared with that expected from the general population. Our findings highlight the need for awareness by survivors and their healthcare providers for potential risk related to SPL.

Original languageEnglish
Pages (from-to)71-83
Number of pages13
JournalEuropean Journal of Cancer
Volume117
DOIs
Publication statusPublished - Aug 2019

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Survivors
Leukemia
Cohort Studies
Neoplasms
Myeloid Leukemia
Confidence Intervals
Incidence
Lymphoid Leukemia
Health Personnel
Population
Odds Ratio
Demography

Keywords

  • Childhood cancer survivors
  • Lymphoid leukaemias
  • Myeloid leukaemias
  • Second cancers
  • Subsequent primary leukaemia

ASJC Scopus subject areas

  • Oncology
  • Cancer Research

Cite this

Risk of subsequent primary leukaemias among 69,460 five-year survivors of childhood cancer diagnosed from 1940 to 2008 in Europe : A cohort study within PanCareSurFup. / Allodji, Rodrigue S.; Hawkins, Mike M.; Bright, Chloe J.; Fidler-Benaoudia, Miranda M.; Winter, David L.; Alessi, Daniela; Fresneau, Brice; Journy, Neige; Morsellino, Vera; Bárdi, Edit; Bautz, Andrea; Byrne, Julianne; Feijen, Elizabeth (Lieke) AM; Teepen, Jop C.; Vu-Bezin, Giao; Rubino, Carole; Garwicz, Stanislaw; Grabow, Desiree; Gudmundsdottir, Thorgerdur; Guha, Joyeeta; Hau, Eva Maria; Jankovic, Momcilo; Kaatsch, Peter; Kaiser, Melanie; Linge, Helena; Muraca, Monica; Llanas, Damien; Veres, Cristina; Øfstaas, Hilde; Diallo, Ibrahima; Mansouri, Imene; Ronckers, Cecile M.; Skinner, Roderick; Terenziani, Monica; Wesenberg, Finn; Wiebe, Thomas; Sacerdote, Carlotta; Jakab, Zsuzsanna; Haupt, Riccardo; Lähteenmäki, Päivi; Zaletel, Lorna Zadravec; Kuehni, Claudia E.; Winther, Jeanette F.; Michel, Gisela; Kremer, Leontien C.M.; Hjorth, Lars; Haddy, Nadia; de Vathaire, Florent; Reulen, Raoul C.

In: European Journal of Cancer, Vol. 117, 08.2019, p. 71-83.

Research output: Contribution to journalArticle

Allodji, RS, Hawkins, MM, Bright, CJ, Fidler-Benaoudia, MM, Winter, DL, Alessi, D, Fresneau, B, Journy, N, Morsellino, V, Bárdi, E, Bautz, A, Byrne, J, Feijen, ELAM, Teepen, JC, Vu-Bezin, G, Rubino, C, Garwicz, S, Grabow, D, Gudmundsdottir, T, Guha, J, Hau, EM, Jankovic, M, Kaatsch, P, Kaiser, M, Linge, H, Muraca, M, Llanas, D, Veres, C, Øfstaas, H, Diallo, I, Mansouri, I, Ronckers, CM, Skinner, R, Terenziani, M, Wesenberg, F, Wiebe, T, Sacerdote, C, Jakab, Z, Haupt, R, Lähteenmäki, P, Zaletel, LZ, Kuehni, CE, Winther, JF, Michel, G, Kremer, LCM, Hjorth, L, Haddy, N, de Vathaire, F & Reulen, RC 2019, 'Risk of subsequent primary leukaemias among 69,460 five-year survivors of childhood cancer diagnosed from 1940 to 2008 in Europe: A cohort study within PanCareSurFup', European Journal of Cancer, vol. 117, pp. 71-83. https://doi.org/10.1016/j.ejca.2019.05.013
Allodji, Rodrigue S. ; Hawkins, Mike M. ; Bright, Chloe J. ; Fidler-Benaoudia, Miranda M. ; Winter, David L. ; Alessi, Daniela ; Fresneau, Brice ; Journy, Neige ; Morsellino, Vera ; Bárdi, Edit ; Bautz, Andrea ; Byrne, Julianne ; Feijen, Elizabeth (Lieke) AM ; Teepen, Jop C. ; Vu-Bezin, Giao ; Rubino, Carole ; Garwicz, Stanislaw ; Grabow, Desiree ; Gudmundsdottir, Thorgerdur ; Guha, Joyeeta ; Hau, Eva Maria ; Jankovic, Momcilo ; Kaatsch, Peter ; Kaiser, Melanie ; Linge, Helena ; Muraca, Monica ; Llanas, Damien ; Veres, Cristina ; Øfstaas, Hilde ; Diallo, Ibrahima ; Mansouri, Imene ; Ronckers, Cecile M. ; Skinner, Roderick ; Terenziani, Monica ; Wesenberg, Finn ; Wiebe, Thomas ; Sacerdote, Carlotta ; Jakab, Zsuzsanna ; Haupt, Riccardo ; Lähteenmäki, Päivi ; Zaletel, Lorna Zadravec ; Kuehni, Claudia E. ; Winther, Jeanette F. ; Michel, Gisela ; Kremer, Leontien C.M. ; Hjorth, Lars ; Haddy, Nadia ; de Vathaire, Florent ; Reulen, Raoul C. / Risk of subsequent primary leukaemias among 69,460 five-year survivors of childhood cancer diagnosed from 1940 to 2008 in Europe : A cohort study within PanCareSurFup. In: European Journal of Cancer. 2019 ; Vol. 117. pp. 71-83.
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title = "Risk of subsequent primary leukaemias among 69,460 five-year survivors of childhood cancer diagnosed from 1940 to 2008 in Europe: A cohort study within PanCareSurFup",
abstract = "Background: Survivors of childhood cancers are at risk of developing subsequent primary leukaemias (SPLs), but the long-term risks beyond 20 years of treatment are still unclear. We investigated the risk of SPLs in five-year childhood cancer survivors using a large-scale pan-European (PanCareSurFup) cohort and evaluated variations in the risk by cancer and demographic factors. Methods: This largest-ever assembled cohort comprises 69,460 five-year childhood cancer survivors from 12 European countries. Standardised incidence ratios (SIRs) and absolute excess risks (AERs) were calculated. Results: One hundred fifteen survivors developed an SPL including 86 myeloid leukaemias (subsequent primary myeloid leukaemias [SPMLs]), 17 lymphoid leukaemias and 12 other types of leukaemias; of these SPLs, 31 (27{\%}) occurred beyond 20 years from the first childhood cancer diagnosis. Compared with the general population, childhood cancer survivors had a fourfold increased risk (SIR = 3.7, 95{\%} confidence interval [CI]: 3.1 to 4.5) of developing leukaemia, and eight leukaemias per 100,000 person-years (AER = 7.5, 95{\%} CI: 6.0 to 9.2) occurred in excess of that expected. The risks remained significantly elevated beyond 20 years from the first primary malignancy (SIR = 2.4, 95{\%} CI: 1.6 to 3.4). Overall, the risk ratio for SPML (SIR = 5.8, 95{\%} CI: 4.6 to 7.1) was higher than that for other SPLs. Conclusions: We demonstrate that beyond 20 years after childhood cancer diagnosis, survivors experience an increased risk for SPLs compared with that expected from the general population. Our findings highlight the need for awareness by survivors and their healthcare providers for potential risk related to SPL.",
keywords = "Childhood cancer survivors, Lymphoid leukaemias, Myeloid leukaemias, Second cancers, Subsequent primary leukaemia",
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journal = "European Journal of Cancer",
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TY - JOUR

T1 - Risk of subsequent primary leukaemias among 69,460 five-year survivors of childhood cancer diagnosed from 1940 to 2008 in Europe

T2 - A cohort study within PanCareSurFup

AU - Allodji, Rodrigue S.

AU - Hawkins, Mike M.

AU - Bright, Chloe J.

AU - Fidler-Benaoudia, Miranda M.

AU - Winter, David L.

AU - Alessi, Daniela

AU - Fresneau, Brice

AU - Journy, Neige

AU - Morsellino, Vera

AU - Bárdi, Edit

AU - Bautz, Andrea

AU - Byrne, Julianne

AU - Feijen, Elizabeth (Lieke) AM

AU - Teepen, Jop C.

AU - Vu-Bezin, Giao

AU - Rubino, Carole

AU - Garwicz, Stanislaw

AU - Grabow, Desiree

AU - Gudmundsdottir, Thorgerdur

AU - Guha, Joyeeta

AU - Hau, Eva Maria

AU - Jankovic, Momcilo

AU - Kaatsch, Peter

AU - Kaiser, Melanie

AU - Linge, Helena

AU - Muraca, Monica

AU - Llanas, Damien

AU - Veres, Cristina

AU - Øfstaas, Hilde

AU - Diallo, Ibrahima

AU - Mansouri, Imene

AU - Ronckers, Cecile M.

AU - Skinner, Roderick

AU - Terenziani, Monica

AU - Wesenberg, Finn

AU - Wiebe, Thomas

AU - Sacerdote, Carlotta

AU - Jakab, Zsuzsanna

AU - Haupt, Riccardo

AU - Lähteenmäki, Päivi

AU - Zaletel, Lorna Zadravec

AU - Kuehni, Claudia E.

AU - Winther, Jeanette F.

AU - Michel, Gisela

AU - Kremer, Leontien C.M.

AU - Hjorth, Lars

AU - Haddy, Nadia

AU - de Vathaire, Florent

AU - Reulen, Raoul C.

PY - 2019/8

Y1 - 2019/8

N2 - Background: Survivors of childhood cancers are at risk of developing subsequent primary leukaemias (SPLs), but the long-term risks beyond 20 years of treatment are still unclear. We investigated the risk of SPLs in five-year childhood cancer survivors using a large-scale pan-European (PanCareSurFup) cohort and evaluated variations in the risk by cancer and demographic factors. Methods: This largest-ever assembled cohort comprises 69,460 five-year childhood cancer survivors from 12 European countries. Standardised incidence ratios (SIRs) and absolute excess risks (AERs) were calculated. Results: One hundred fifteen survivors developed an SPL including 86 myeloid leukaemias (subsequent primary myeloid leukaemias [SPMLs]), 17 lymphoid leukaemias and 12 other types of leukaemias; of these SPLs, 31 (27%) occurred beyond 20 years from the first childhood cancer diagnosis. Compared with the general population, childhood cancer survivors had a fourfold increased risk (SIR = 3.7, 95% confidence interval [CI]: 3.1 to 4.5) of developing leukaemia, and eight leukaemias per 100,000 person-years (AER = 7.5, 95% CI: 6.0 to 9.2) occurred in excess of that expected. The risks remained significantly elevated beyond 20 years from the first primary malignancy (SIR = 2.4, 95% CI: 1.6 to 3.4). Overall, the risk ratio for SPML (SIR = 5.8, 95% CI: 4.6 to 7.1) was higher than that for other SPLs. Conclusions: We demonstrate that beyond 20 years after childhood cancer diagnosis, survivors experience an increased risk for SPLs compared with that expected from the general population. Our findings highlight the need for awareness by survivors and their healthcare providers for potential risk related to SPL.

AB - Background: Survivors of childhood cancers are at risk of developing subsequent primary leukaemias (SPLs), but the long-term risks beyond 20 years of treatment are still unclear. We investigated the risk of SPLs in five-year childhood cancer survivors using a large-scale pan-European (PanCareSurFup) cohort and evaluated variations in the risk by cancer and demographic factors. Methods: This largest-ever assembled cohort comprises 69,460 five-year childhood cancer survivors from 12 European countries. Standardised incidence ratios (SIRs) and absolute excess risks (AERs) were calculated. Results: One hundred fifteen survivors developed an SPL including 86 myeloid leukaemias (subsequent primary myeloid leukaemias [SPMLs]), 17 lymphoid leukaemias and 12 other types of leukaemias; of these SPLs, 31 (27%) occurred beyond 20 years from the first childhood cancer diagnosis. Compared with the general population, childhood cancer survivors had a fourfold increased risk (SIR = 3.7, 95% confidence interval [CI]: 3.1 to 4.5) of developing leukaemia, and eight leukaemias per 100,000 person-years (AER = 7.5, 95% CI: 6.0 to 9.2) occurred in excess of that expected. The risks remained significantly elevated beyond 20 years from the first primary malignancy (SIR = 2.4, 95% CI: 1.6 to 3.4). Overall, the risk ratio for SPML (SIR = 5.8, 95% CI: 4.6 to 7.1) was higher than that for other SPLs. Conclusions: We demonstrate that beyond 20 years after childhood cancer diagnosis, survivors experience an increased risk for SPLs compared with that expected from the general population. Our findings highlight the need for awareness by survivors and their healthcare providers for potential risk related to SPL.

KW - Childhood cancer survivors

KW - Lymphoid leukaemias

KW - Myeloid leukaemias

KW - Second cancers

KW - Subsequent primary leukaemia

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DO - 10.1016/j.ejca.2019.05.013

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JO - European Journal of Cancer

JF - European Journal of Cancer

SN - 0959-8049

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