Risk factors for mortality in infancy and childhood in children with major congenital anomalies: A European population‐based cohort study

Author:

Tan Joachim1ORCID,Glinianaia Svetlana V.2ORCID,Rankin Judith2ORCID,Pierini Anna3,Santoro Michele3,Coi Alessio3,Garne Ester4,Loane Maria5,Given Joanne E.5ORCID,Brigden Joanna1,Ballardini Elisa6,Cavero‐Carbonell Clara7,de Walle Hermien E. K.8,García‐Villodre Laura7,Gatt Miriam9,Gissler Mika101112,Heino Anna10,Jordan Sue13,Khoshnood Babak14ORCID,Klungsoyr Kari1516,Lelong Nathalie14,Lutke Renée L.8,Neville Amanda J.17,Tucker David18,Urhoj Stine K.419,Wellesley Diana20,Morris Joan K.1ORCID

Affiliation:

1. Population Health Research Institute St George's University of London London UK

2. Population Health Sciences Institute Newcastle University Newcastle upon Tyne UK

3. Unit of Epidemiology of Rare Diseases and Congenital Anomalies Institute of Clinical Physiology, National Research Council Pisa Italy

4. Department of Paediatrics and Adolescent Medicine, Lillebaelt Hospital University Hospital of Southern Denmark Kolding Denmark

5. Faculty of Life and Health Sciences Ulster University Coleraine UK

6. Neonatal Intensive Care Unit, Paediatric Section, IMER Registry (Emilia Romagna Registry of Birth Defects), Department of Medical Sciences University of Ferrara Ferrara Italy

7. Rare Diseases Research Unit Foundation for the Promotion of Health and Biomedical Research in the Valencian Region Valencia Spain

8. Department of Genetics University of Groningen, University Medical Center Groningen Groningen The Netherlands

9. Malta Congenital Anomalies Registry Directorate for Health Information and Research Tal‐Pietà Malta

10. Department of Knowledge Brokers THL Finnish Institute for Health and Welfare Helsinki Finland

11. Region Stockholm Academic Primary Health Care Centre Stockholm Sweden

12. Department of Molecular Medicine and Surgery Karolinska Institutet Stockholm Sweden

13. Faculty of Medicine, Health and Life Science Swansea University Swansea UK

14. Université Paris Cité, Centre of Research in Epidemiology and StatisticS (CRESS) Obstetrical Perinatal and Pediatric Epidemiology Research Team (EPOPé) Paris France

15. Department of Global Public Health and Primary Care University of Bergen Bergen Norway

16. Division of Mental and Physical Health Norwegian Institute of Public Health Bergen Norway

17. IMER Registry (Emilia Romagna Registry of Birth Defects), Center for Clinical and Epidemiological Research University of Ferrara and Azienda Ospedaliero Universitaria di Ferrara Ferrara Italy

18. Congenital Anomaly Register & Information Service for Wales (CARIS) Public Health Knowledge and Research, Public Health Wales Swansea UK

19. Section of Epidemiology, Department of Public Health University of Copenhagen Copenhagen Denmark

20. University of Southampton and Wessex Clinical Genetics Service, Princess Anne Hospital Southampton UK

Abstract

AbstractBackgroundPreterm birth and young maternal age are known risk factors for infant and childhood mortality. There is limited knowledge of the impact of these risk factors in children born with major congenital anomalies (CAs), who have inherently higher risks of death compared with other children.ObjectivesTo investigate the risk factors for mortality up to age 10 years in children born with specific major CAs.MethodsThis population‐based cohort study involved 150,198 livebirths from 1995 to 2014 in 13 European CA registries linked to mortality data. Cox proportional hazards models estimated the association of gestational age, maternal age and child's sex with death <1 year and 1–9 years for the whole cohort and by CA subgroup. Hazard ratios (HR) from each registry were pooled using multivariate meta‐analysis.ResultsPreterm birth had a dose–response association with mortality; compared with infants born at 37+ weeks gestation, those born at <28, 28–31 and 32–36 weeks had 14.88 (95% CI 12.57, 17.62), 8.39 (95% CI 7.16, 9.85) and 3.88 (95% CI 3.40, 4.43) times higher risk of death <1 year, respectively. The corresponding risks at 1–9 years were 4.99 (95% CI 2.94, 8.48), 3.09 (95% CI 2.28, 4.18) and 2.04 (95% CI 1.69, 2.46) times higher, respectively. Maternal age <20 years (versus 20–34 years) was a risk factor for death <1 year (HR 1.30, 95% CI 1.09, 1.54) and 1–9 years (HR 1.58, 95% CI 1.19, 2.10). Females had 1.22 (95% CI 1.07, 1.39) times higher risk of death between 1 and 9 years than males.ConclusionPreterm birth was associated with considerably higher infant and childhood mortality in children with CAs, comparable to estimates reported elsewhere for the background population. Additional risk factors included young maternal age and female sex. Information on risk factors could benefit clinical care and guide counselling of parents following CA diagnoses.

Funder

Horizon 2020 Framework Programme

Publisher

Wiley

Subject

Pediatrics, Perinatology and Child Health,Epidemiology

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