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

Maria D Chirlaque; Rafael Peris-Bonet; Antonia Sánchez; Ofelia Cruz; Rafael Marcos-Gragera; Gonzalo Gutiérrez-Ávila; José R Quirós-García; Fernando Almela-Vich; Arantza López de Munain; Maria J Sánchez; Paula Franch-Sureda; Eva Ardanaz; Jaume Galceran; Carmen Martos; Diego Salmerón; Gemma Gatta; Laura Botta; Adela Cañete; The Spanish Childhood Cancer Epidemiology Working Group

doi:10.3390/cancers15245889

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

Cancers (Basel). 2023 Dec 18;15(24):5889. doi: 10.3390/cancers15245889.

Authors

Maria D Chirlaque^{1

2

3}, Rafael Peris-Bonet⁴, Antonia Sánchez², Ofelia Cruz⁵, Rafael Marcos-Gragera^{1

6

7

8

9}, Gonzalo Gutiérrez-Ávila¹⁰, José R Quirós-García¹¹, Fernando Almela-Vich¹², Arantza López de Munain¹³, Maria J Sánchez^{1

14

15}, Paula Franch-Sureda¹⁶, Eva Ardanaz^{1

17

18}, Jaume Galceran^{19

20

21}, Carmen Martos²², Diego Salmerón^{1

3

23}, Gemma Gatta²⁴, Laura Botta²⁴, Adela Cañete^{4

25

26}, The Spanish Childhood Cancer Epidemiology Working Group

Affiliations

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.

Keywords: Spain; adolescent CNS tumours; childhood CNS tumours; incidence; paediatric oncology; population-based cancer registries; survival; time trends.

Abstract

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