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Cancer Epidemiology, Biomarkers & Prevention
Cancer Epidemiology, Biomarkers & Prevention
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Risk Factors of Subsequent Central Nervous System Tumors after Childhood and Adolescent Cancers: Findings from the French Childhood Cancer Survivor Study

Neige Marie Yvanne Journy, Wael Salem Zrafi, Stéphanie Bolle, Brice Fresneau, Claire Alapetite, Rodrigue Setcheou Allodji, Delphine Berchery, Nadia Haddy, Isao Kobayashi, Martine Labbé, Hélène Pacquement, Claire Pluchart, Boris Schwartz, Vincent Souchard, Cécile Thomas-Teinturier, Cristina Veres, Giao Vu-Bezin, Ibrahima Diallo and Florent de Vathaire
Neige Marie Yvanne Journy
1Inserm U1018, CESP, Radiation Epidemiology Team, Gustave Roussy, Villejuif, Paris, France.
2Paris-Saclay, Paris-Sud University, Villejuif, Paris, France.
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  • For correspondence: neige.journy@gustaveroussy.fr
Wael Salem Zrafi
1Inserm U1018, CESP, Radiation Epidemiology Team, Gustave Roussy, Villejuif, Paris, France.
2Paris-Saclay, Paris-Sud University, Villejuif, Paris, France.
3Department of Radiation Oncology, Gustave Roussy, Villejuif, Paris, France.
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  • ORCID record for Wael Salem Zrafi
Stéphanie Bolle
3Department of Radiation Oncology, Gustave Roussy, Villejuif, Paris, France.
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  • ORCID record for Stéphanie Bolle
Brice Fresneau
1Inserm U1018, CESP, Radiation Epidemiology Team, Gustave Roussy, Villejuif, Paris, France.
2Paris-Saclay, Paris-Sud University, Villejuif, Paris, France.
4Department of Children and Adolescent Oncology, Gustave Roussy, Villejuif, Paris, France.
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Claire Alapetite
5Department of Radiation Oncology, Institut Curie, Paris, France.
6Institut Curie, Proton Therapy Center, Orsay, Paris, France.
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Rodrigue Setcheou Allodji
1Inserm U1018, CESP, Radiation Epidemiology Team, Gustave Roussy, Villejuif, Paris, France.
2Paris-Saclay, Paris-Sud University, Villejuif, Paris, France.
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  • ORCID record for Rodrigue Setcheou Allodji
Delphine Berchery
7Department of Medical Information, Institut Claudius Régaud, Toulouse, France.
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Nadia Haddy
1Inserm U1018, CESP, Radiation Epidemiology Team, Gustave Roussy, Villejuif, Paris, France.
2Paris-Saclay, Paris-Sud University, Villejuif, Paris, France.
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Isao Kobayashi
1Inserm U1018, CESP, Radiation Epidemiology Team, Gustave Roussy, Villejuif, Paris, France.
2Paris-Saclay, Paris-Sud University, Villejuif, Paris, France.
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Martine Labbé
1Inserm U1018, CESP, Radiation Epidemiology Team, Gustave Roussy, Villejuif, Paris, France.
2Paris-Saclay, Paris-Sud University, Villejuif, Paris, France.
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Hélène Pacquement
8Department of Pediatric Medical Oncology, Institut Curie, Paris, France.
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Claire Pluchart
9Department of Pediatric Hematology-Oncology, Institut Jean Godinot, Reims, France.
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Boris Schwartz
1Inserm U1018, CESP, Radiation Epidemiology Team, Gustave Roussy, Villejuif, Paris, France.
2Paris-Saclay, Paris-Sud University, Villejuif, Paris, France.
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Vincent Souchard
1Inserm U1018, CESP, Radiation Epidemiology Team, Gustave Roussy, Villejuif, Paris, France.
2Paris-Saclay, Paris-Sud University, Villejuif, Paris, France.
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Cécile Thomas-Teinturier
1Inserm U1018, CESP, Radiation Epidemiology Team, Gustave Roussy, Villejuif, Paris, France.
2Paris-Saclay, Paris-Sud University, Villejuif, Paris, France.
10Department of Paediatric Endocrinology, APHP, Paris-Sud Hospital, site Bicêtre, Le Kremlin Bicêtre, France.
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Cristina Veres
1Inserm U1018, CESP, Radiation Epidemiology Team, Gustave Roussy, Villejuif, Paris, France.
2Paris-Saclay, Paris-Sud University, Villejuif, Paris, France.
11Inserm UMR 1030, Gustave Roussy Cancer Campus, Villejuif, Paris, France.
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Giao Vu-Bezin
1Inserm U1018, CESP, Radiation Epidemiology Team, Gustave Roussy, Villejuif, Paris, France.
2Paris-Saclay, Paris-Sud University, Villejuif, Paris, France.
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Ibrahima Diallo
1Inserm U1018, CESP, Radiation Epidemiology Team, Gustave Roussy, Villejuif, Paris, France.
2Paris-Saclay, Paris-Sud University, Villejuif, Paris, France.
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Florent de Vathaire
1Inserm U1018, CESP, Radiation Epidemiology Team, Gustave Roussy, Villejuif, Paris, France.
2Paris-Saclay, Paris-Sud University, Villejuif, Paris, France.
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DOI: 10.1158/1055-9965.EPI-20-0735 Published January 2021
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Abstract

Background: Childhood or adolescent cancer survivors are at increased risks of subsequent primary neoplasms (SPN) of the central nervous system (CNS) after cranial irradiation. In a large multicentric cohort, we investigated clinical and therapeutic factors associated with the long-term risk of CNS SPN, and quantified the dose–response relationships.

Methods: We selected all CNS SPN cases diagnosed up to 2016 among members of the French Childhood Cancer Survivor Study at least 5 years after first cancer diagnosis in 1946–2000. Four controls per case were randomly selected within the cohort and matched by sex, year of/age at first cancer diagnosis, and follow-up time. On the basis of medical and radiological reports, cumulative radiation doses received to the SPN or matched location were retrospectively estimated using mathematical phantoms. We computed conditional logistic regression models.

Results: Meningioma risk significantly increased with higher radiation doses [excess OR per Gy (EOR/Gy) = 1.377; P < 0.001; 86 cases; median latency time = 30 years], after adjustment for reported genetic syndromes and first CNS tumor. It was higher among youngest individuals at first cancer diagnosis, but did not vary with follow-up time. On the opposite, radiation-related glioma risk (EOR/Gy = 0.049; P = 0.11; 47 cases; median latency time = 17 years) decreased over time (P for time effect = 0.05). There was a significant association between meningioma risk and cumulative doses of alkylating agents, but no association with growth hormone therapy.

Conclusions: The surveillance of patients with cranial irradiation should continue beyond 30 years after treatment.

Impact: The identified risk factors may inform long-term surveillance strategies.

Footnotes

  • Note: Supplementary data for this article are available at Cancer Epidemiology, Biomarkers & Prevention Online (http://cebp.aacrjournals.org/).

  • Cancer Epidemiol Biomarkers Prev 2021;30:133–41

  • Received May 14, 2020.
  • Revision received July 31, 2020.
  • Accepted September 30, 2020.
  • Published first October 8, 2020.
  • ©2020 American Association for Cancer Research.
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Cancer Epidemiology Biomarkers & Prevention: 30 (1)
January 2021
Volume 30, Issue 1
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Risk Factors of Subsequent Central Nervous System Tumors after Childhood and Adolescent Cancers: Findings from the French Childhood Cancer Survivor Study
Neige Marie Yvanne Journy, Wael Salem Zrafi, Stéphanie Bolle, Brice Fresneau, Claire Alapetite, Rodrigue Setcheou Allodji, Delphine Berchery, Nadia Haddy, Isao Kobayashi, Martine Labbé, Hélène Pacquement, Claire Pluchart, Boris Schwartz, Vincent Souchard, Cécile Thomas-Teinturier, Cristina Veres, Giao Vu-Bezin, Ibrahima Diallo and Florent de Vathaire
Cancer Epidemiol Biomarkers Prev January 1 2021 (30) (1) 133-141; DOI: 10.1158/1055-9965.EPI-20-0735

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Risk Factors of Subsequent Central Nervous System Tumors after Childhood and Adolescent Cancers: Findings from the French Childhood Cancer Survivor Study
Neige Marie Yvanne Journy, Wael Salem Zrafi, Stéphanie Bolle, Brice Fresneau, Claire Alapetite, Rodrigue Setcheou Allodji, Delphine Berchery, Nadia Haddy, Isao Kobayashi, Martine Labbé, Hélène Pacquement, Claire Pluchart, Boris Schwartz, Vincent Souchard, Cécile Thomas-Teinturier, Cristina Veres, Giao Vu-Bezin, Ibrahima Diallo and Florent de Vathaire
Cancer Epidemiol Biomarkers Prev January 1 2021 (30) (1) 133-141; DOI: 10.1158/1055-9965.EPI-20-0735
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