Risk of subsequent primary oral cancer in a cohort of 69,460 5-year survivors of childhood and adolescent cancer in Europe: the PanCareSurFup study

Ceren Sunguc; Michael Hawkins; David L. Winter; Isabelle M. Dudley; Emma J. Heymer; Jop C. Teepen; Rodrigue S. Allodji; Fabiën N. Belle; Francesca Bagnasco; Julianne Byrne; Edit Bárdi; Cécile M. Ronckers; Nadia Haddy; Thorgerdur Gudmundsdottir; Stanislaw Garwicz; Momcilo Jankovic; Helena J. H. van der Pal; Maja Česen Mazić; Christina Schindera; Desiree Grabow; Milena M. Maule; Peter Kaatsch; Brice Fresneau; Gisela Michel; Roderick Skinner; Thomas Wiebe; Carlotta Sacerdote; Zsuzsanna Jakab; Maria Winther Gunnes; Monica Terenziani; Jeanette F. Winther; Päivi M. Lähteenmäki; Lorna Zadravec Zaletel; Claudia E. Kuehni; Leontien C. Kremer; Riccardo Haupt; Florent de Vathaire; Lars Hjorth; Raoul Reulen

doi:10.1038/s41416-022-02016-w

Risk of subsequent primary oral cancer in a cohort of 69,460 5-year survivors of childhood and adolescent cancer in Europe: the PanCareSurFup study

Ceren Sunguc, Michael Hawkins, David L. Winter, Isabelle M. Dudley, Emma J. Heymer, Jop C. Teepen, Rodrigue S. Allodji, Fabiën N. Belle, Francesca Bagnasco, Julianne Byrne, Edit Bárdi, Cécile M. Ronckers, Nadia Haddy, Thorgerdur Gudmundsdottir, Stanislaw Garwicz, Momcilo Jankovic, Helena J. H. van der Pal, Maja Česen Mazić, Christina Schindera, Desiree GrabowMilena M. Maule, Peter Kaatsch, Brice Fresneau, Gisela Michel, Roderick Skinner, Thomas Wiebe, Carlotta Sacerdote, Zsuzsanna Jakab, Maria Winther Gunnes, Monica Terenziani, Jeanette F. Winther, Päivi M. Lähteenmäki, Lorna Zadravec Zaletel, Claudia E. Kuehni, Leontien C. Kremer, Riccardo Haupt, Florent de Vathaire, Lars Hjorth, Raoul Reulen^*

^*Corresponding author for this work

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Abstract

Background: Survivors of childhood cancer are at risk of subsequent primary malignant neoplasms (SPNs), but the risk for rarer types of SPNs, such as oral cancer, is uncertain. Previous studies included few oral SPNs, hence large-scale cohorts are required to identify groups at risks.

Methods: The PanCareSurFup cohort includes 69,460 5-year survivors of childhood cancer across Europe. Risks of oral SPNs were defined by standardised incidence ratios (SIRs), absolute excess risks and cumulative incidence.

Results: One hundred and forty-five oral SPNs (64 salivary gland, 38 tongue, 20 pharynx, 2 lip, and 21 other) were ascertained among 143 survivors. Survivors were at 5-fold risk of an oral SPN (95% CI: 4.4–5.6). Survivors of leukaemia were at greatest risk (SIR = 19.2; 95% CI: 14.6–25.2) followed by bone sarcoma (SIR = 6.4, 95% CI: 3.7–11.0), Hodgkin lymphoma (SIR = 6.2, 95% CI: 3.9–9.9) and soft-tissue sarcoma (SIR = 5.0, 95% CI: 3.0–8.5). Survivors treated with radiotherapy were at 33-fold risk of salivary gland SPNs (95% CI: 25.3–44.5), particularly Hodgkin lymphoma (SIR = 66.2, 95% CI: 43.6–100.5) and leukaemia (SIR = 50.5, 95% CI: 36.1–70.7) survivors. Survivors treated with chemotherapy had a substantially increased risk of a tongue SPN (SIR = 15.9, 95% CI: 10.6–23.7).

Conclusions: Previous radiotherapy increases the risk of salivary gland SPNs considerably, while chemotherapy increases the risk of tongue SPNs substantially. Awareness of these risks among both health-care professionals and survivors could play a crucial role in detecting oral SPNs early.

Original language	English
Pages (from-to)	80-90
Number of pages	11
Journal	British Journal of Cancer
Volume	128
Issue number	1
Early online date	1 Nov 2022
DOIs	https://doi.org/10.1038/s41416-022-02016-w
Publication status	Published - 26 Jan 2023

Bibliographical note

Keywords

Humans
Adolescent
Hodgkin Disease
Risk Factors
Neoplasms, Second Primary/epidemiology
Survivors
Sarcoma
Europe/epidemiology
Bone Neoplasms/complications
Leukemia/epidemiology
Incidence
Mouth Neoplasms/epidemiology

UN SDGs

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

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10.1038/s41416-022-02016-wLicence: Creative Commons: Attribution (CC BY)

SungucC2022RiskFinal published version, 843 KBLicence: Creative Commons: Attribution (CC BY)

FP7 COLLAB - PanCareSurFup: PanCare Childhood and Adolescent Cancer Survivor Care and Follow-up Studies.
Hawkins, M.
European Commission - Management Costs, European Commission
1/02/11 → 31/01/17
Project: Research

Cite this

Sunguc, C., Hawkins, M., Winter, D. L., Dudley, I. M., Heymer, E. J., Teepen, J. C., Allodji, R. S., Belle, F. N., Bagnasco, F., Byrne, J., Bárdi, E., Ronckers, C. M., Haddy, N., Gudmundsdottir, T., Garwicz, S., Jankovic, M., Pal, H. J. H. V. D., Mazić, M. Č., Schindera, C., ... Reulen, R. (2023). Risk of subsequent primary oral cancer in a cohort of 69,460 5-year survivors of childhood and adolescent cancer in Europe: the PanCareSurFup study. British Journal of Cancer, 128(1), 80-90. https://doi.org/10.1038/s41416-022-02016-w

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title = "Risk of subsequent primary oral cancer in a cohort of 69,460 5-year survivors of childhood and adolescent cancer in Europe: the PanCareSurFup study",

abstract = "Background: Survivors of childhood cancer are at risk of subsequent primary malignant neoplasms (SPNs), but the risk for rarer types of SPNs, such as oral cancer, is uncertain. Previous studies included few oral SPNs, hence large-scale cohorts are required to identify groups at risks.Methods: The PanCareSurFup cohort includes 69,460 5-year survivors of childhood cancer across Europe. Risks of oral SPNs were defined by standardised incidence ratios (SIRs), absolute excess risks and cumulative incidence.Results: One hundred and forty-five oral SPNs (64 salivary gland, 38 tongue, 20 pharynx, 2 lip, and 21 other) were ascertained among 143 survivors. Survivors were at 5-fold risk of an oral SPN (95% CI: 4.4–5.6). Survivors of leukaemia were at greatest risk (SIR = 19.2; 95% CI: 14.6–25.2) followed by bone sarcoma (SIR = 6.4, 95% CI: 3.7–11.0), Hodgkin lymphoma (SIR = 6.2, 95% CI: 3.9–9.9) and soft-tissue sarcoma (SIR = 5.0, 95% CI: 3.0–8.5). Survivors treated with radiotherapy were at 33-fold risk of salivary gland SPNs (95% CI: 25.3–44.5), particularly Hodgkin lymphoma (SIR = 66.2, 95% CI: 43.6–100.5) and leukaemia (SIR = 50.5, 95% CI: 36.1–70.7) survivors. Survivors treated with chemotherapy had a substantially increased risk of a tongue SPN (SIR = 15.9, 95% CI: 10.6–23.7).Conclusions: Previous radiotherapy increases the risk of salivary gland SPNs considerably, while chemotherapy increases the risk of tongue SPNs substantially. Awareness of these risks among both health-care professionals and survivors could play a crucial role in detecting oral SPNs early.",

keywords = "Humans, Adolescent, Hodgkin Disease, Risk Factors, Neoplasms, Second Primary/epidemiology, Survivors, Sarcoma, Europe/epidemiology, Bone Neoplasms/complications, Leukemia/epidemiology, Incidence, Mouth Neoplasms/epidemiology",

author = "Ceren Sunguc and Michael Hawkins and Winter, {David L.} and Dudley, {Isabelle M.} and Heymer, {Emma J.} and Teepen, {Jop C.} and Allodji, {Rodrigue S.} and Belle, {Fabi{\"e}n N.} and Francesca Bagnasco and Julianne Byrne and Edit B{\'a}rdi and Ronckers, {C{\'e}cile M.} and Nadia Haddy and Thorgerdur Gudmundsdottir and Stanislaw Garwicz and Momcilo Jankovic and Pal, {Helena J. H. van der} and Mazi{\'c}, {Maja {\v C}esen} and Christina Schindera and Desiree Grabow and Maule, {Milena M.} and Peter Kaatsch and Brice Fresneau and Gisela Michel and Roderick Skinner and Thomas Wiebe and Carlotta Sacerdote and Zsuzsanna Jakab and Gunnes, {Maria Winther} and Monica Terenziani and Winther, {Jeanette F.} and L{\"a}hteenm{\"a}ki, {P{\"a}ivi M.} and Zaletel, {Lorna Zadravec} and Kuehni, {Claudia E.} and Kremer, {Leontien C.} and Riccardo Haupt and Vathaire, {Florent de} and Lars Hjorth and Raoul Reulen",

note = "{\textcopyright} 2022. The Author(s).",

year = "2023",

month = jan,

day = "26",

doi = "10.1038/s41416-022-02016-w",

language = "English",

volume = "128",

pages = "80--90",

journal = "British Journal of Cancer",

issn = "0007-0920",

publisher = "Cancer Research UK",

number = "1",

}

Sunguc, C, Hawkins, M, Winter, DL, Dudley, IM, Heymer, EJ, Teepen, JC, Allodji, RS, Belle, FN, Bagnasco, F, Byrne, J, Bárdi, E, Ronckers, CM, Haddy, N, Gudmundsdottir, T, Garwicz, S, Jankovic, M, Pal, HJHVD, Mazić, MČ, Schindera, C, Grabow, D, Maule, MM, Kaatsch, P, Fresneau, B, Michel, G, Skinner, R, Wiebe, T, Sacerdote, C, Jakab, Z, Gunnes, MW, Terenziani, M, Winther, JF, Lähteenmäki, PM, Zaletel, LZ, Kuehni, CE, Kremer, LC, Haupt, R, Vathaire, FD, Hjorth, L & Reulen, R 2023, 'Risk of subsequent primary oral cancer in a cohort of 69,460 5-year survivors of childhood and adolescent cancer in Europe: the PanCareSurFup study', British Journal of Cancer, vol. 128, no. 1, pp. 80-90. https://doi.org/10.1038/s41416-022-02016-w

TY - JOUR

T1 - Risk of subsequent primary oral cancer in a cohort of 69,460 5-year survivors of childhood and adolescent cancer in Europe

T2 - the PanCareSurFup study

AU - Sunguc, Ceren

AU - Hawkins, Michael

AU - Winter, David L.

AU - Dudley, Isabelle M.

AU - Heymer, Emma J.

AU - Teepen, Jop C.

AU - Allodji, Rodrigue S.

AU - Belle, Fabiën N.

AU - Bagnasco, Francesca

AU - Byrne, Julianne

AU - Bárdi, Edit

AU - Ronckers, Cécile M.

AU - Haddy, Nadia

AU - Gudmundsdottir, Thorgerdur

AU - Garwicz, Stanislaw

AU - Jankovic, Momcilo

AU - Pal, Helena J. H. van der

AU - Mazić, Maja Česen

AU - Schindera, Christina

AU - Grabow, Desiree

AU - Maule, Milena M.

AU - Kaatsch, Peter

AU - Fresneau, Brice

AU - Michel, Gisela

AU - Skinner, Roderick

AU - Wiebe, Thomas

AU - Sacerdote, Carlotta

AU - Jakab, Zsuzsanna

AU - Gunnes, Maria Winther

AU - Terenziani, Monica

AU - Winther, Jeanette F.

AU - Lähteenmäki, Päivi M.

AU - Zaletel, Lorna Zadravec

AU - Kuehni, Claudia E.

AU - Kremer, Leontien C.

AU - Haupt, Riccardo

AU - Vathaire, Florent de

AU - Hjorth, Lars

AU - Reulen, Raoul

PY - 2023/1/26

Y1 - 2023/1/26

N2 - Background: Survivors of childhood cancer are at risk of subsequent primary malignant neoplasms (SPNs), but the risk for rarer types of SPNs, such as oral cancer, is uncertain. Previous studies included few oral SPNs, hence large-scale cohorts are required to identify groups at risks.Methods: The PanCareSurFup cohort includes 69,460 5-year survivors of childhood cancer across Europe. Risks of oral SPNs were defined by standardised incidence ratios (SIRs), absolute excess risks and cumulative incidence.Results: One hundred and forty-five oral SPNs (64 salivary gland, 38 tongue, 20 pharynx, 2 lip, and 21 other) were ascertained among 143 survivors. Survivors were at 5-fold risk of an oral SPN (95% CI: 4.4–5.6). Survivors of leukaemia were at greatest risk (SIR = 19.2; 95% CI: 14.6–25.2) followed by bone sarcoma (SIR = 6.4, 95% CI: 3.7–11.0), Hodgkin lymphoma (SIR = 6.2, 95% CI: 3.9–9.9) and soft-tissue sarcoma (SIR = 5.0, 95% CI: 3.0–8.5). Survivors treated with radiotherapy were at 33-fold risk of salivary gland SPNs (95% CI: 25.3–44.5), particularly Hodgkin lymphoma (SIR = 66.2, 95% CI: 43.6–100.5) and leukaemia (SIR = 50.5, 95% CI: 36.1–70.7) survivors. Survivors treated with chemotherapy had a substantially increased risk of a tongue SPN (SIR = 15.9, 95% CI: 10.6–23.7).Conclusions: Previous radiotherapy increases the risk of salivary gland SPNs considerably, while chemotherapy increases the risk of tongue SPNs substantially. Awareness of these risks among both health-care professionals and survivors could play a crucial role in detecting oral SPNs early.

AB - Background: Survivors of childhood cancer are at risk of subsequent primary malignant neoplasms (SPNs), but the risk for rarer types of SPNs, such as oral cancer, is uncertain. Previous studies included few oral SPNs, hence large-scale cohorts are required to identify groups at risks.Methods: The PanCareSurFup cohort includes 69,460 5-year survivors of childhood cancer across Europe. Risks of oral SPNs were defined by standardised incidence ratios (SIRs), absolute excess risks and cumulative incidence.Results: One hundred and forty-five oral SPNs (64 salivary gland, 38 tongue, 20 pharynx, 2 lip, and 21 other) were ascertained among 143 survivors. Survivors were at 5-fold risk of an oral SPN (95% CI: 4.4–5.6). Survivors of leukaemia were at greatest risk (SIR = 19.2; 95% CI: 14.6–25.2) followed by bone sarcoma (SIR = 6.4, 95% CI: 3.7–11.0), Hodgkin lymphoma (SIR = 6.2, 95% CI: 3.9–9.9) and soft-tissue sarcoma (SIR = 5.0, 95% CI: 3.0–8.5). Survivors treated with radiotherapy were at 33-fold risk of salivary gland SPNs (95% CI: 25.3–44.5), particularly Hodgkin lymphoma (SIR = 66.2, 95% CI: 43.6–100.5) and leukaemia (SIR = 50.5, 95% CI: 36.1–70.7) survivors. Survivors treated with chemotherapy had a substantially increased risk of a tongue SPN (SIR = 15.9, 95% CI: 10.6–23.7).Conclusions: Previous radiotherapy increases the risk of salivary gland SPNs considerably, while chemotherapy increases the risk of tongue SPNs substantially. Awareness of these risks among both health-care professionals and survivors could play a crucial role in detecting oral SPNs early.

KW - Humans

KW - Adolescent

KW - Hodgkin Disease

KW - Risk Factors

KW - Neoplasms, Second Primary/epidemiology

KW - Survivors

KW - Sarcoma

KW - Europe/epidemiology

KW - Bone Neoplasms/complications

KW - Leukemia/epidemiology

KW - Incidence

KW - Mouth Neoplasms/epidemiology

U2 - 10.1038/s41416-022-02016-w

DO - 10.1038/s41416-022-02016-w

M3 - Article

C2 - 36319851

SN - 0007-0920

VL - 128

SP - 80

EP - 90

JO - British Journal of Cancer

JF - British Journal of Cancer

IS - 1

ER -

Risk of subsequent primary oral cancer in a cohort of 69,460 5-year survivors of childhood and adolescent cancer in Europe: the PanCareSurFup study

Abstract

Bibliographical note

Keywords

UN SDGs

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Projects

FP7 COLLAB - PanCareSurFup: PanCare Childhood and Adolescent Cancer Survivor Care and Follow-up Studies.

Cite this