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 Guha; Eva-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

doi:10.1016/j.ejca.2019.05.013

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

Applied Health Research

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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 language	English
Pages (from-to)	71-83
Number of pages	13
Journal	European Journal of Cancer
Volume	117
Early online date	28 Jun 2019
DOIs	https://doi.org/10.1016/j.ejca.2019.05.013
Publication status	Published - Aug 2019

Bibliographical note

Keywords

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

UN SDGs

This output contributes to the following UN Sustainable Development Goals (SDGs)

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Rodrigue_S_Allodji_et_al_Risk_of_subsequent_primary_leukaemias_among_69,460_five-year_survivors_of_childhood_cancer_European_Journal_of_Cancer_2019
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Accepted author manuscript, 29.3 KBLicence: Creative Commons: Attribution-NonCommercial-NoDerivs (CC BY-NC-ND)

Cite this

Allodji, R. S., Hawkins, M. M., Bright, C. J., Fidler-Benaoudia, M. M., Winter, D. L., Alessi, D., Fresneau, B., Journy, N., Morsellino, V., Bárdi, E., Bautz, A., Byrne, J., Feijen, E. L. A., Teepen, J. C., Vu-Bezin, G., Rubino, C., Garwicz, S., Grabow, D., Gudmundsdottir, T., ... Reulen, R. C. (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, 117, 71-83. Advance online publication. https://doi.org/10.1016/j.ejca.2019.05.013

@article{e82e1325c37c4e97b7ee4f245506caaf,

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, Second cancers, Subsequent primary leukaemia, Myeloid leukaemias, Lymphoid leukaemias",

author = "Allodji, {Rodrigue S} and Hawkins, {Mike M} and Bright, {Chloe J} and Fidler-Benaoudia, {Miranda M} and Winter, {David L} and Daniela Alessi and Brice Fresneau and Neige Journy and Vera Morsellino and Edit B{\'a}rdi and Andrea Bautz and Julianne Byrne and Feijen, {Elizabeth Lieke Am} and Teepen, {Jop C} and Giao Vu-Bezin and Carole Rubino and Stanislaw Garwicz and Desiree Grabow and Thorgerdur Gudmundsdottir and Joyeeta Guha and Eva-Maria Hau and Momcilo Jankovic and Peter Kaatsch and Melanie Kaiser and Helena Linge and Monica Muraca and Damien Llanas and Cristina Veres and Hilde {\O}fstaas and Ibrahima Diallo and Imene Mansouri and Ronckers, {Cecile M} and Roderick Skinner and Monica Terenziani and Finn Wesenberg and Thomas Wiebe and Carlotta Sacerdote and Zsuzsanna Jakab and Riccardo Haupt and P{\"a}ivi L{\"a}hteenm{\"a}ki and Zaletel, {Lorna Zadravec} and Kuehni, {Claudia E} and Winther, {Jeanette F} and Gisela Michel and Kremer, {Leontien C M} and Lars Hjorth and Nadia Haddy and {de Vathaire}, Florent and Reulen, {Raoul C}",

year = "2019",

month = aug,

doi = "10.1016/j.ejca.2019.05.013",

language = "English",

volume = "117",

pages = "71--83",

journal = "European Journal of Cancer",

issn = "0959-8049",

publisher = "Elsevier",

}

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, ELA, Teepen, JC, Vu-Bezin, G, Rubino, C, Garwicz, S, Grabow, D, Gudmundsdottir, T, Guha, J, Hau, E-M, 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

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 - Second cancers

KW - Subsequent primary leukaemia

KW - Myeloid leukaemias

KW - Lymphoid leukaemias

U2 - 10.1016/j.ejca.2019.05.013

DO - 10.1016/j.ejca.2019.05.013

M3 - Article

C2 - 31260818

SN - 0959-8049

VL - 117

SP - 71

EP - 83

JO - European Journal of Cancer

JF - European Journal of Cancer

ER -

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

Bibliographical note

Keywords

UN SDGs

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