Childhood and Adolescent Central Nervous System Tumours in Spain: Incidence and Survival over 20 Years: A Historical Baseline for Current Assessment

Author:

Chirlaque Maria D.123,Peris-Bonet Rafael4ORCID,Sánchez Antonia2,Cruz Ofelia5,Marcos-Gragera Rafael16789ORCID,Gutiérrez-Ávila Gonzalo10,Quirós-García José R.11,Almela-Vich Fernando12,López de Munain Arantza13ORCID,Sánchez Maria J.11415ORCID,Franch-Sureda Paula16,Ardanaz Eva11718,Galceran Jaume192021,Martos Carmen22,Salmerón Diego1323ORCID,Gatta Gemma24,Botta Laura24,Cañete Adela42526,

Affiliation:

1. Consortium for Biomedical Research in Epidemiology and Public Health (CIBERESP), 28029 Madrid, Spain

2. Department of Epidemiology, Murcia Regional Health Authority, 30071 Murcia, Spain

3. Department of Epidemiology, Regional Health Council, IMIB-Arrixaca, Murcia University, El Palmar, 30120 Murcia, Spain

4. Spanish Registry of Childhood Tumours (RETI-SEHOP), University of Valencia, Faculty of Medicine, 46010 Valencia, Spain

5. Neuro-Oncology Unit, Paediatric Cancer Centre, Sant Joan de Déu Hospital, Esplugues de Llobregat, 08950 Barcelona, Spain

6. Epidemiology Unit and Girona Cancer Registry, Oncology Co-Ordination Plan, Catalonian Oncology Institute, 17004 Girona, Spain

7. Josep Trueta Girona Biomedical Research Institute (IDIBGI), Salt, 17190 Girona, Spain

8. Statistics, Econometrics and Health Research Group (GRECS), University of Girona, 17004 Girona, Spain

9. Josep Carreras Leukaemia Research Institute, 08916 Badalona, Spain

10. Castile-La Mancha Cancer Registry, Regional Health Authority, 45071 Toledo, Spain

11. Asturias Cancer Registry, Public Health Directorate, 33006 Oviedo Asturias, Spain

12. Valencian Regional Childhood Cancer Registry, Non-Communicable Disease Epidemiology and Surveillance Department, General Subdirectorate of Epidemiology and Health Surveillance, General Directorate of Public Health and Addictions, Regional Public Health Authority, Valencian Regional Authority, 46010 Valencia, Spain

13. Basque Country Cancer Registry, Health Department, Basque Country Regional Authority, 01010 Vitoria-Gasteiz, Spain

14. Andalusian School of Public Health (EASP), 18011 Granada, Spain

15. Granada Bio-Health Research Institute, 18012 Granada, Spain

16. Mallorca Cancer Registry, General Directorate of Public Health and Participation, Balearic Isles Health Research Institute (IdISBa), 07010 Palma de Mallorca, Spain

17. Navarre Public Health Institute, 31003 Pamplona, Spain

18. IdiSNA, Navarre Health Research Institute, 31008 Pamplona, Spain

19. Tarragona Cancer Registry, Cancer Epidemiology and Prevention Service, Sant Joan de Reus University Teaching Hospital, 43204 Reus, Spain

20. Pere Virgili Health Research Institute (IISPV), 43204 Reus, Spain

21. Faculty of Medicine and Health Sciences, Rovira i Virgili University (URV), 43003 Reus, Spain

22. Rare Diseases Research Unit, Foundation for the Promotion of Health and Biomedical Research in the Valencian Region (FISABIO), 46020 Valencia, Spain

23. Department of Health and Social Sciences, University of Murcia, 30100 Murcia, Spain

24. Evaluative Epidemiology Unit, Department of Epidemiology and Data Science, Fondazione IRCCS, Istituto Nazionale dei Tumori, 20133 Milan, Italy

25. Paediatric Oncology Department, La Fe Hospital, 46026 Valencia, Spain

26. Paediatrics, Obstetrics and Gynaecology Department, Faculty of Medicine, University of Valencia, 46010 Valencia, Spain

Abstract

Background: Central nervous system (CNS) neoplasms are highly frequent solid tumours in children and adolescents. While some studies have shown a rise in their incidence in Europe, others have not. Survival remains limited. We addressed two questions about these tumours in Spain: (1) Is incidence increasing? and (2) Has survival improved? Methods: This population-based study included 1635 children and 328 adolescents from 11 population-based cancer registries with International Classification of Childhood Cancer Group III tumours, incident in 1983–2007. Age-specific and age-standardised (world population) incidence rates (ASRws) were calculated. Incidence time trends were characterised using annual percent change (APC) obtained with Joinpoint. Cases from 1991 to 2005 (1171) were included in Kaplan–Meier survival analyses, and the results were evaluated with log-rank and log-rank for trend tests. Children’s survival was age-standardised using: (1) the age distribution of cases and the corresponding trends assessed with Joinpoint; and (2) European weights for comparison with Europe. Results: ASRw 1983–2007: children: 32.7 cases/106; adolescents: 23.5 cases/106. The overall incidence of all tumours increased across 1983–2007 in children and adolescents. Considering change points, the APCs were: (1) children: 1983–1993, 4.3%^ (1.1; 7.7); 1993–2007, −0.2% (−1.9; 1.6); (2) adolescents: 1983–2004: 2.9%^ (0.9; 4.9); 2004–2007: −7.7% (−40; 41.9). For malignant tumours, the trends were not significant. 5-year survival was 65% (1991–2005), with no significant trends (except for non-malignant tumours). Conclusions: CNS tumour incidence in Spain was found to be similar to that in Europe. Rises in incidence may be mostly attributable to changes in the registration of non-malignant tumours. The overall malignant CNS tumour trend was compatible with reports for Southern Europe. Survival was lower than in Europe, without improvement over time. We provide a baseline for assessing current paediatric oncology achievements and incidence in respect of childhood and adolescent CNS tumours.

Funder

Carlos III Institute of Health

RTICC

Publisher

MDPI AG

Subject

Cancer Research,Oncology

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