Estimating Global Burden of Disease due to congenital anomaly: an analysis of European data.

Helen Dolk, Breidge Boyle, Marie-Claude Addor, Larraitz Arriola, Ingeborg Barisic, Fabrizio Bianchi, Melinda Csaky-Szunyogh, HEK de Walle, Carlos Matias Dias, E Draper, Miriam Gatt, Garne Ester, Martin Haeusler, Karin Kallen, Anna Latos-Bielenska , B McDonnell, Carmel Mullaney, Vera Nelen, Amanda Neville, Mary O'Mahony & 11 others A Queisser-Wahrendorf, Hanitra Randrianaivo-Ranjatoelina, J Rankin, Anke Rissmann, Annukka Ritvanen, Catherine Rounding, David Tucker, Christine Verellen-Dumoulin, Diana Wellesley, B Wreyford, Natalya Zymak-Zakutnia

Research output: Contribution to journalArticle

14 Citations (Scopus)

Abstract

OBJECTIVE: To validate the estimates of Global Burden of Disease (GBD) due to congenital anomaly for Europe by comparing infant mortality data collected by EUROCAT registries with the WHO Mortality Database, and by assessing the significance of stillbirths and terminations of pregnancy for fetal anomaly (TOPFA) in the interpretation of infant mortality statistics. DESIGN, SETTING AND OUTCOME MEASURES: EUROCAT is a network of congenital anomaly registries collecting data on live births, fetal deaths from 20 weeks' gestation and TOPFA. Data from 29 registries in 19 countries were analysed for 2005-2009, and infant mortality (deaths of live births at age <1 year) compared with the WHO Mortality Database. Eight EUROCAT countries were excluded from further analysis on the basis that this comparison showed poor ascertainment of survival status. RESULTS: According to WHO, 17%-42% of infant mortality was attributed to congenital anomaly. In 11 EUROCAT countries, average infant mortality with congenital anomaly was 1.1 per 1000 births, with higher rates where TOPFA is illegal (Malta 3.0, Ireland 2.1). The rate of stillbirths with congenital anomaly was 0.6 per 1000. The average TOPFA prevalence was 4.6 per 1000, nearly three times more prevalent than stillbirths and infant deaths combined. TOPFA also impacted on the prevalence of postneonatal survivors with non-lethal congenital anomaly. CONCLUSIONS: By excluding TOPFA and stillbirths from GBD years of life lost (YLL) estimates, GBD underestimates the burden of disease due to congenital anomaly, and thus declining YLL over time may obscure lack of progress in primary, secondary and tertiary prevention.
LanguageEnglish
PagesF22-F28
JournalArchives of Disease in Childhood: Fetal and Neonatal Edition
Volume103
Issue number1
DOIs
Publication statusPublished - 30 Jun 2017

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Infant Mortality
Stillbirth
Pregnancy
Registries
Live Birth
Malta
Tertiary Prevention
Databases
Fetal Death
Mortality
Pregnancy Rate
Primary Prevention
Secondary Prevention
Ireland
Survivors
Global Burden of Disease
Outcome Assessment (Health Care)
Parturition
Survival
Infant Death

Keywords

  • Congenital Anomaly
  • mortality

Cite this

Dolk, Helen ; Boyle, Breidge ; Addor, Marie-Claude ; Arriola, Larraitz ; Barisic, Ingeborg ; Bianchi, Fabrizio ; Csaky-Szunyogh, Melinda ; de Walle, HEK ; Dias, Carlos Matias ; Draper, E ; Gatt, Miriam ; Ester, Garne ; Haeusler, Martin ; Kallen, Karin ; Latos-Bielenska , Anna ; McDonnell, B ; Mullaney, Carmel ; Nelen, Vera ; Neville, Amanda ; O'Mahony, Mary ; Queisser-Wahrendorf, A ; Randrianaivo-Ranjatoelina, Hanitra ; Rankin, J ; Rissmann, Anke ; Ritvanen, Annukka ; Rounding, Catherine ; Tucker, David ; Verellen-Dumoulin, Christine ; Wellesley, Diana ; Wreyford, B ; Zymak-Zakutnia, Natalya. / Estimating Global Burden of Disease due to congenital anomaly: an analysis of European data. In: Archives of Disease in Childhood: Fetal and Neonatal Edition. 2017 ; Vol. 103, No. 1. pp. F22-F28.
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abstract = "OBJECTIVE: To validate the estimates of Global Burden of Disease (GBD) due to congenital anomaly for Europe by comparing infant mortality data collected by EUROCAT registries with the WHO Mortality Database, and by assessing the significance of stillbirths and terminations of pregnancy for fetal anomaly (TOPFA) in the interpretation of infant mortality statistics. DESIGN, SETTING AND OUTCOME MEASURES: EUROCAT is a network of congenital anomaly registries collecting data on live births, fetal deaths from 20 weeks' gestation and TOPFA. Data from 29 registries in 19 countries were analysed for 2005-2009, and infant mortality (deaths of live births at age <1 year) compared with the WHO Mortality Database. Eight EUROCAT countries were excluded from further analysis on the basis that this comparison showed poor ascertainment of survival status. RESULTS: According to WHO, 17{\%}-42{\%} of infant mortality was attributed to congenital anomaly. In 11 EUROCAT countries, average infant mortality with congenital anomaly was 1.1 per 1000 births, with higher rates where TOPFA is illegal (Malta 3.0, Ireland 2.1). The rate of stillbirths with congenital anomaly was 0.6 per 1000. The average TOPFA prevalence was 4.6 per 1000, nearly three times more prevalent than stillbirths and infant deaths combined. TOPFA also impacted on the prevalence of postneonatal survivors with non-lethal congenital anomaly. CONCLUSIONS: By excluding TOPFA and stillbirths from GBD years of life lost (YLL) estimates, GBD underestimates the burden of disease due to congenital anomaly, and thus declining YLL over time may obscure lack of progress in primary, secondary and tertiary prevention.",
keywords = "Congenital Anomaly, mortality",
author = "Helen Dolk and Breidge Boyle and Marie-Claude Addor and Larraitz Arriola and Ingeborg Barisic and Fabrizio Bianchi and Melinda Csaky-Szunyogh and {de Walle}, HEK and Dias, {Carlos Matias} and E Draper and Miriam Gatt and Garne Ester and Martin Haeusler and Karin Kallen and Anna Latos-Bielenska and B McDonnell and Carmel Mullaney and Vera Nelen and Amanda Neville and Mary O'Mahony and A Queisser-Wahrendorf and Hanitra Randrianaivo-Ranjatoelina and J Rankin and Anke Rissmann and Annukka Ritvanen and Catherine Rounding and David Tucker and Christine Verellen-Dumoulin and Diana Wellesley and B Wreyford and Natalya Zymak-Zakutnia",
year = "2017",
month = "6",
day = "30",
doi = "10.1136/archdischild-2016-311845",
language = "English",
volume = "103",
pages = "F22--F28",
journal = "Archives of Disease in Childhood: Fetal and Neonatal Edition",
issn = "1359-2998",
number = "1",

}

Dolk, H, Boyle, B, Addor, M-C, Arriola, L, Barisic, I, Bianchi, F, Csaky-Szunyogh, M, de Walle, HEK, Dias, CM, Draper, E, Gatt, M, Ester, G, Haeusler, M, Kallen, K, Latos-Bielenska , A, McDonnell, B, Mullaney, C, Nelen, V, Neville, A, O'Mahony, M, Queisser-Wahrendorf, A, Randrianaivo-Ranjatoelina, H, Rankin, J, Rissmann, A, Ritvanen, A, Rounding, C, Tucker, D, Verellen-Dumoulin, C, Wellesley, D, Wreyford, B & Zymak-Zakutnia, N 2017, 'Estimating Global Burden of Disease due to congenital anomaly: an analysis of European data.', Archives of Disease in Childhood: Fetal and Neonatal Edition, vol. 103, no. 1, pp. F22-F28. https://doi.org/10.1136/archdischild-2016-311845

Estimating Global Burden of Disease due to congenital anomaly: an analysis of European data. / Dolk, Helen; Boyle, Breidge; Addor, Marie-Claude; Arriola, Larraitz; Barisic, Ingeborg ; Bianchi, Fabrizio; Csaky-Szunyogh, Melinda; de Walle, HEK; Dias, Carlos Matias; Draper, E; Gatt, Miriam; Ester, Garne; Haeusler, Martin; Kallen, Karin; Latos-Bielenska , Anna; McDonnell, B; Mullaney, Carmel; Nelen, Vera; Neville, Amanda; O'Mahony, Mary; Queisser-Wahrendorf, A; Randrianaivo-Ranjatoelina, Hanitra; Rankin, J; Rissmann, Anke; Ritvanen, Annukka; Rounding, Catherine; Tucker, David; Verellen-Dumoulin, Christine; Wellesley, Diana; Wreyford, B; Zymak-Zakutnia, Natalya.

In: Archives of Disease in Childhood: Fetal and Neonatal Edition, Vol. 103, No. 1, 30.06.2017, p. F22-F28.

Research output: Contribution to journalArticle

TY - JOUR

T1 - Estimating Global Burden of Disease due to congenital anomaly: an analysis of European data.

AU - Dolk, Helen

AU - Boyle, Breidge

AU - Addor, Marie-Claude

AU - Arriola, Larraitz

AU - Barisic, Ingeborg

AU - Bianchi, Fabrizio

AU - Csaky-Szunyogh, Melinda

AU - de Walle, HEK

AU - Dias, Carlos Matias

AU - Draper, E

AU - Gatt, Miriam

AU - Ester, Garne

AU - Haeusler, Martin

AU - Kallen, Karin

AU - Latos-Bielenska , Anna

AU - McDonnell, B

AU - Mullaney, Carmel

AU - Nelen, Vera

AU - Neville, Amanda

AU - O'Mahony, Mary

AU - Queisser-Wahrendorf, A

AU - Randrianaivo-Ranjatoelina, Hanitra

AU - Rankin, J

AU - Rissmann, Anke

AU - Ritvanen, Annukka

AU - Rounding, Catherine

AU - Tucker, David

AU - Verellen-Dumoulin, Christine

AU - Wellesley, Diana

AU - Wreyford, B

AU - Zymak-Zakutnia, Natalya

PY - 2017/6/30

Y1 - 2017/6/30

N2 - OBJECTIVE: To validate the estimates of Global Burden of Disease (GBD) due to congenital anomaly for Europe by comparing infant mortality data collected by EUROCAT registries with the WHO Mortality Database, and by assessing the significance of stillbirths and terminations of pregnancy for fetal anomaly (TOPFA) in the interpretation of infant mortality statistics. DESIGN, SETTING AND OUTCOME MEASURES: EUROCAT is a network of congenital anomaly registries collecting data on live births, fetal deaths from 20 weeks' gestation and TOPFA. Data from 29 registries in 19 countries were analysed for 2005-2009, and infant mortality (deaths of live births at age <1 year) compared with the WHO Mortality Database. Eight EUROCAT countries were excluded from further analysis on the basis that this comparison showed poor ascertainment of survival status. RESULTS: According to WHO, 17%-42% of infant mortality was attributed to congenital anomaly. In 11 EUROCAT countries, average infant mortality with congenital anomaly was 1.1 per 1000 births, with higher rates where TOPFA is illegal (Malta 3.0, Ireland 2.1). The rate of stillbirths with congenital anomaly was 0.6 per 1000. The average TOPFA prevalence was 4.6 per 1000, nearly three times more prevalent than stillbirths and infant deaths combined. TOPFA also impacted on the prevalence of postneonatal survivors with non-lethal congenital anomaly. CONCLUSIONS: By excluding TOPFA and stillbirths from GBD years of life lost (YLL) estimates, GBD underestimates the burden of disease due to congenital anomaly, and thus declining YLL over time may obscure lack of progress in primary, secondary and tertiary prevention.

AB - OBJECTIVE: To validate the estimates of Global Burden of Disease (GBD) due to congenital anomaly for Europe by comparing infant mortality data collected by EUROCAT registries with the WHO Mortality Database, and by assessing the significance of stillbirths and terminations of pregnancy for fetal anomaly (TOPFA) in the interpretation of infant mortality statistics. DESIGN, SETTING AND OUTCOME MEASURES: EUROCAT is a network of congenital anomaly registries collecting data on live births, fetal deaths from 20 weeks' gestation and TOPFA. Data from 29 registries in 19 countries were analysed for 2005-2009, and infant mortality (deaths of live births at age <1 year) compared with the WHO Mortality Database. Eight EUROCAT countries were excluded from further analysis on the basis that this comparison showed poor ascertainment of survival status. RESULTS: According to WHO, 17%-42% of infant mortality was attributed to congenital anomaly. In 11 EUROCAT countries, average infant mortality with congenital anomaly was 1.1 per 1000 births, with higher rates where TOPFA is illegal (Malta 3.0, Ireland 2.1). The rate of stillbirths with congenital anomaly was 0.6 per 1000. The average TOPFA prevalence was 4.6 per 1000, nearly three times more prevalent than stillbirths and infant deaths combined. TOPFA also impacted on the prevalence of postneonatal survivors with non-lethal congenital anomaly. CONCLUSIONS: By excluding TOPFA and stillbirths from GBD years of life lost (YLL) estimates, GBD underestimates the burden of disease due to congenital anomaly, and thus declining YLL over time may obscure lack of progress in primary, secondary and tertiary prevention.

KW - Congenital Anomaly

KW - mortality

U2 - 10.1136/archdischild-2016-311845

DO - 10.1136/archdischild-2016-311845

M3 - Article

VL - 103

SP - F22-F28

JO - Archives of Disease in Childhood: Fetal and Neonatal Edition

T2 - Archives of Disease in Childhood: Fetal and Neonatal Edition

JF - Archives of Disease in Childhood: Fetal and Neonatal Edition

SN - 1359-2998

IS - 1

ER -