Causes of death in children with congenital anomalies up to age 10 in eight European countries.


Journal

BMJ paediatrics open
ISSN: 2399-9772
Titre abrégé: BMJ Paediatr Open
Pays: England
ID NLM: 101715309

Informations de publication

Date de publication:
06 2023
Historique:
received: 22 07 2022
accepted: 20 08 2022
medline: 26 6 2023
pubmed: 24 6 2023
entrez: 23 6 2023
Statut: ppublish

Résumé

Congenital anomalies (CAs) increase the risk of death during infancy and childhood. This study aimed to evaluate the accuracy of using death certificates to estimate the burden of CAs on mortality for children under 10 years old. Children born alive with a major CA between 1 January 1995 and 31 December 2014, from 13 population-based European CA registries were linked to mortality records up to their 10th birthday or 31 December 2015, whichever was earlier. In total 4199 neonatal, 2100 postneonatal and 1087 deaths in children aged 1-9 years were reported. The underlying cause of death was a CA in 71% (95% CI 64% to 78%) of neonatal and 68% (95% CI 61% to 74%) of postneonatal infant deaths. For neonatal deaths the proportions varied by registry from 45% to 89% and by anomaly from 53% for Down syndrome to 94% for tetralogy of Fallot. In children aged 1-9, 49% (95% CI 42% to 57%) were attributed to a CA. Comparing mortality in children with anomalies to population mortality predicts that over 90% of all deaths at all ages are attributable to the anomalies. The specific CA was often not reported on the death certificate, even for lethal anomalies such as trisomy 13 (only 80% included the code for trisomy 13). Data on the underlying cause of death from death certificates alone are not sufficient to evaluate the burden of CAs on infant and childhood mortality across countries and over time. Linked data from CA registries and death certificates are necessary for obtaining accurate estimates.

Sections du résumé

BACKGROUND
Congenital anomalies (CAs) increase the risk of death during infancy and childhood. This study aimed to evaluate the accuracy of using death certificates to estimate the burden of CAs on mortality for children under 10 years old.
METHODS
Children born alive with a major CA between 1 January 1995 and 31 December 2014, from 13 population-based European CA registries were linked to mortality records up to their 10th birthday or 31 December 2015, whichever was earlier.
RESULTS
In total 4199 neonatal, 2100 postneonatal and 1087 deaths in children aged 1-9 years were reported. The underlying cause of death was a CA in 71% (95% CI 64% to 78%) of neonatal and 68% (95% CI 61% to 74%) of postneonatal infant deaths. For neonatal deaths the proportions varied by registry from 45% to 89% and by anomaly from 53% for Down syndrome to 94% for tetralogy of Fallot. In children aged 1-9, 49% (95% CI 42% to 57%) were attributed to a CA. Comparing mortality in children with anomalies to population mortality predicts that over 90% of all deaths at all ages are attributable to the anomalies. The specific CA was often not reported on the death certificate, even for lethal anomalies such as trisomy 13 (only 80% included the code for trisomy 13).
CONCLUSIONS
Data on the underlying cause of death from death certificates alone are not sufficient to evaluate the burden of CAs on infant and childhood mortality across countries and over time. Linked data from CA registries and death certificates are necessary for obtaining accurate estimates.

Identifiants

pubmed: 37353235
pii: 10.1136/bmjpo-2022-001617
doi: 10.1136/bmjpo-2022-001617
pmc: PMC10367017
pii:
doi:

Types de publication

Journal Article Research Support, Non-U.S. Gov't

Langues

eng

Sous-ensembles de citation

IM

Informations de copyright

© Author(s) (or their employer(s)) 2023. Re-use permitted under CC BY-NC. No commercial re-use. See rights and permissions. Published by BMJ.

Déclaration de conflit d'intérêts

Competing interests: None.

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Auteurs

Anke Rissmann (A)

Malformation Monitoring Centre Saxony-Anhalt, Medical Faculty, Otto von Guericke Universitat Magdeburg, Magdeburg, Germany.

Joachim Tan (J)

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

Svetlana V Glinianaia (SV)

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

Judith Rankin (J)

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

Anna Pierini (A)

Fondazione Toscana Gabriele Monasterio, Pisa, Italy.
Unit of Epidemiology of Rare Diseases and Congenital Anomalies, Institute of Clinical Physiology National Research Council, Pisa, Italy.

Michele Santoro (M)

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

Alessio Coi (A)

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

Ester Garne (E)

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

Maria Loane (M)

Centre for Maternal, Fetal and Infant Research, Institute of Nursing and Health Research, Ulster University, Belfast, UK.

Joanne Given (J)

Centre for Maternal, Fetal and Infant Research, Institute of Nursing and Health Research, Ulster University, Belfast, UK.

Abigail Reid (A)

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

Amaia Aizpurua (A)

Health Division of Gipuzkoa, Biodonostia Health Research Institute, Donostia-San Sebastian, Spain.

Diana Akhmedzhanova (D)

OMNI-Net for Children International Charitable Fund, Rivne Regional Medical Diagnostic Center, Rivne, Ukraine.

Elisa Ballardini (E)

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

Ingeborg Barisic (I)

Centre of Excellence for Reproductive and Regenerative Medicine, Children's Hospital Zagreb, Medical School University of Zagreb, Zagreb, Croatia.

Clara Cavero-Carbonell (C)

Rare Diseases Research Join Unit, Foundation for the Promotion of Health and Biomedical Research and Universitat de Valencia, Valencia, Spain.

Hermien E K de Walle (HEK)

Department of Genetics, Groningen University, Groningen, The Netherlands.

Miriam Gatt (M)

Malta Congenital Anomalies Register, Directorate for Health Information and Research, Tal-Pietà, Malta.

Mika Gissler (M)

Department of Knowledge Brokers, THL Finnish Institute for Health and Welfare, Helsinki, Finland.
Department of Molecular Medicine and Surgery, Karolinska Institutet, Stockholm, Sweden.

Anna Heino (A)

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

Sue Jordan (S)

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

Stine Kjaer Urhoj (SK)

Department of Paediatrics and Adolescent Medicine, Lillebaelt Hospital - University Hospital of Southern Denmark, Kolding, Denmark.
Department of Public Health, University of Copenhagen, Kobenhavn, Denmark.

Kari Klungsøyr (K)

Department of Global Public Health and Primary Care, University of Bergen, Bergen, Norway.
Medical Birth Registry, Nasjonalt folkehelseinstitutt, Bergen, Norway.

Renee Lutke (R)

Department of Genetics, Groningen University, Groningen, The Netherlands.

Olatz Mokoroa (O)

Health Division of Gipuzkoa, Biodonostia Health Research Institute, Donostia-San Sebastian, Spain.

Amanda Julie Neville (AJ)

IMER Registry, Centre for Clinical and Epidemiological Research, University of Ferrara and Azienda Ospedaliero Universitario di Ferrara, Ferrara, Italy.

Daniel S Thayer (DS)

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

Diana G Wellesley (DG)

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

Lyubov Yevtushok (L)

OMNI-Net for Children International Charitable Fund, Rivne Regional Medical Diagnostic Center, Rivne, Ukraine.

Oscar Zurriaga (O)

Rare Diseases Research Join Unit, Foundation for the Promotion of Health and Biomedical Research and Universitat de Valencia, Valencia, Spain.
CIBER de Epidemiología y Salud Pública, Instituto de Salud Carlos III, Madrid, Spain.

Joan Morris (J)

Population Health Research Institute, St George's University of London, London, UK jmorris@sgul.ac.uk.

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