Cancer Causes & Control

, Volume 30, Issue 10, pp 1075–1085 | Cite as

Family history of cancer and the risk of childhood brain tumors: a pooled analysis of the ESCALE and ESTELLE studies (SFCE)

  • Nicolas Vidart d’Egurbide BagazgoïtiaEmail author
  • Helen D. Bailey
  • Laurent Orsi
  • Léa Guerrini-Rousseau
  • Anne-Isabelle Bertozzi
  • Cécile Faure-Conter
  • Pierre Leblond
  • Isabelle Pellier
  • Claire Freycon
  • François Doz
  • Stéphanie Puget
  • Stéphane Ducassou
  • Brigitte Lacour
  • Jacqueline Clavel
Original Paper



Although some specific genetic syndromes such as neurofibromatosis (NF) have been identified as risk factor of childhood brain tumors (CBT), the potential role of inherited susceptibility in CBT has yet to be elucidated.


To further investigate this, we conducted a pooled analysis of two nationwide case–control studies ESCALE and ESTELLE. The mothers of 509 CBT cases and 3,102 controls aged under 15 years who resided in France at diagnosis/interview, frequency-matched by age and gender, responded to a telephone interview conducted by trained interviewers. Pooled odds ratio (OR) and 95% confidence intervals (95% CI) were estimated using unconditional logistic regression.


CBT was significantly associated with the family history of cancer in relatives (OR 1.2, 95% CI 1.0–1.5). The OR was slightly higher for maternal relatives than for paternal relatives, and when at least two relatives had a history of cancer. CBT was significantly associated with a family history of brain tumor (OR 2.1, 95% CI 1.3–3.7). This association seemed stronger for first-degree relatives (mother, father, and siblings), for whom, by contrast, no association was seen for cancers other than CBT. No specificity by CBT subtypes or by age of the children were found for any of these findings.


Our findings support the hypothesis of a familial susceptibility of CBT, not due to being a known NF carrier.


Childhood cancer Brain tumors Family history of cancer Family history of brain tumor Risk factors Case–control studies France 



Childhood brain tumors


Confidence interval


Constitutional mismatch repair deficiency


Etude Sur les Cancers et les Leucémies de l'Enfant (Study of childhood cancers and leukemias)


Etude Sur les Tumeurs Embryonnaires, Leucémies et Lymphomes de l’Enfant (Study of childhood embryonal tumors, leukemias and lymphomas)


International Classification of Childhood Cancer 3rd Edition




Odds ratio


Registre National des Cancers de l’Enfant (National Registry of Childhood Cancers)


Société Française de lutte contre les Cancers et les leucémies de l’Enfant et de l’Adolescent (French Society for the fight against cancers and leukemia in childhood and adolescence)



The authors would also like to thank all of the Société Française de lutte contre les Cancers de l’Enfant et de l’Adolescent (SFCE) principal investigators: Claire Berger (Centre Hospitalier Universitaire, Saint-Etienne), Christophe Bergeron (Centre Léon Bérard, Lyon), Jean-Louis Bernard (Hôpital La Timone, Marseille), Yves Bertrand (Hôpital Debrousse, Lyon), Pierre Bordigoni (Centre Hospitalier Universitaire, Nancy), Patrick Boutard (Centre Hospitalier Régional Universitaire, Caen), Pascal Chastagner (Centre Hospitalier Universitaire, Nancy), Philippe Colombat (Centre Gatien de Clocheville, Tours), Gérard Couillault (Hôpital d’Enfants, Dijon), Anne-Sophie Defachelles (Oscar Lambret Comprehensive Cancer Center, Lille), François Demeocq (Hôpital Hôtel-Dieu, Clermont-Ferrand), Jean Michon (Institut Curie, Paris), Alain Fischer (Hôpital des Enfants Malades, Paris), Virginie Gandemer (Centre Hospitalier Universitaire – Hôpital Sud, Rennes), Stéphanie Haouy (Hôpital Arnaud de Villeneuve, Montpellier), Jean-Pierre Lamagnere (Centre Gatien de Clocheville, Tours), Françoise Lapierre (Centre Hospitalier Universitaire Jean Bernard, Poitiers), Patrick Lutz (Hôpital de Hautepierre, Strasbourg), Geneviève Margueritte (Hôpital Arnaud de Villeneuve, Montpellier), Françoise Mechinaud (Hôpital Mère et Enfants, Nantes), Gérard Michel (Hôpital La Timone, Marseille), Frédéric Millot (Centre Hospitalier Universitaire Jean Bernard, Poitiers), Philippe le Moine (Hôpital Morvan, Brest), Martine Münzer (American Memorial Hospital, Reims), Brigitte Nelken (Hôpital Jeanne de Flandre, Lille), Brigitte Pautard (Centre Hospitalier Universitaire, Amiens), Yves Perel (Hôpital Pellegrin Tripode, Bordeaux), Alain Pierre-Kahn (Hôpital Enfants Malades, Paris), Christophe Piguet (Centre Hospitalier Régional Universitaire, Limoges), Dominique Plantaz (Centre Hospitalier Universitaire, Grenoble), Emmanuel Plouvier (Centre Hospitalier Régional, Besançon), Marilyne Poiree (Fondation Lenval, Nice), Xavier Rialland (Centre Hospitalier Universitaire, Angers), Alain Robert (Hôpital des Enfants, Toulouse), Hervé Rubie (Hôpital des Enfants, Toulouse), Christian Sainte Rose (Centre Hospitalier Universitaire Necker, Paris), Nicolas Sirvent (Hôpital Arnaud de Villeneuve, Montpellier), Christine Soler (Fondation Lenval, Nice), Dominique Valteau-Couanet (Gustave Roussy, Villejuif), and Jean-Pierre Vannier (Hôpital Charles Nicolle, Rouen). The authors thank all families for their generous participation.


The ESCALE and ESTELLE studies were supported by Grants from INSERM, the Ligue National Contre le Cancer (LNCC), the Fondation de France, the Agence Française de Sécurité Sanitaire des Produits de Santé (AFSSAPS), the Agence Française de Sécurité Sanitaire de l’Environnement et du Travail (AFSSET), the Association pour la Recherche sur le Cancer (ARC), the Agence Française de Sécurité Sanitaire des Produits de Santé (ANSM), the Agence Française de Sécurité Sanitaire de l’Environnement et du Travail (ANSES), the association Cent pour sang la vie, the Institut National du Cancer (INCa), and the Agence Nationale de la Recherche (ANR), and Cancéropôle Ile de France. Nicolas Vidart d’Egurbide Bagazgoïtia’s PhD scholarship is supported by the Institut National du Cancer (INCa_11338).

Compliance with ethical standards

Conflict of interest

The authors declare that they have no conflict of interest.


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Copyright information

© Springer Nature Switzerland AG 2019

Authors and Affiliations

  1. 1.CRESS, UMR1153, Inserm, Paris Descartes UniversityParisFrance
  2. 2.Telethon Kids InstituteThe University of Western AustraliaPerthAustralia
  3. 3.Gustave Roussy, Département de cancérologie de l’enfant et de L’adolescentVillejuifFrance
  4. 4.Unité d’Hémato-Immuno-Oncologie pédiatrique, Pôle Pédiatrique, CHU ToulouseToulouseFrance
  5. 5.Institut d’hématologie et d’oncologie pédiatrique, IHOPe, Centre Léon BérardLyonFrance
  6. 6.Pediatric Oncology UnitOscar Lambret Comprehensive Cancer CenterLilleFrance
  7. 7.Unité immuno-hémato-oncopédiatrie, CHU d’AngersAngersFrance
  8. 8.Clinique de pédiatrie, Hôpital Couple Enfant, CHU Grenoble-AlpesGrenobleFrance
  9. 9.Département de Pédiatrie -Adolescents Et Jeunes Adultes, Institut CurieEt Université Paris DescartesParisFrance
  10. 10.Service de Neurochirurgie pédiatrique, Hôpital Necker-Enfants Malades, Université Paris Descartes, Sorbonne Paris CitéParisFrance
  11. 11.Service d’onco-hématologie pédiatrique, Hôpital Pellegrin TripodeBordeauxFrance
  12. 12.RNCE - National Registry of Childhood Cancers, InsermVillejuif and CHU de NancyFrance

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