Linking a European cohort of children born with congenital anomalies to vital statistics and mortality records: a EUROlinkCAT study

Maria Loane; Joanne Given; Joachim Tan; Abigail Reid; Diana Akhmedzhanova; Gianni Astolfi; Ingeborg Barišić; Nathalie Bertille; LB Bonet; Clara Cavero-Carbonell; O  Mokoroa Carollo; Alessio Coi; J Densem; E Draper; E Garne; M Gatt; SV Glinianaia; A Heino; E Den Hond; S Jordan; B Khoshnood; S Kiuru-Kuhlefelt; K Klungsoyr; N Lelong; L. Renée Lutke; AJ Neville; L Ostapchuk; A Puccini; A Rissmann; M Santoro; I Scanlon; G Thys; D Tucker; SK Urhoj; HEK de Walle; D Wellesley; O Zurriaga; JK Morris

doi:10.1371/journal.pone.0256535

Linking a European cohort of children born with congenital anomalies to vital statistics and mortality records: a EUROlinkCAT study

Maria Loane, Joanne Given, Joachim Tan, Abigail Reid, Diana Akhmedzhanova, Gianni Astolfi, Ingeborg Barišić, Nathalie Bertille, LB Bonet, Clara Cavero-Carbonell, O Mokoroa Carollo, Alessio Coi, J Densem, E Draper, E Garne, M Gatt, SV Glinianaia, A Heino, E Den Hond, S JordanB Khoshnood, S Kiuru-Kuhlefelt, K Klungsoyr, N Lelong, L. Renée Lutke, AJ Neville, L Ostapchuk, A Puccini, A Rissmann, M Santoro, I Scanlon, G Thys, D Tucker, SK Urhoj, HEK de Walle, D Wellesley, O Zurriaga, JK Morris

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Abstract

EUROCAT is a European network of population-based congenital anomaly (CA)
registries. Twenty-one registries agreed to participate in the EUROlinkCAT study to determine if reliable information on the survival of children born with a major CA between 1995 and 2014 can be obtained through linkage to national vital statistics or mortality records. Live birth children with a CA could be linked using personal identifiers to either their national vital statistics (including birth records, death records, hospital records) or to mortality records only, depending on the data available within each region. In total, 18 of 21 registries with data on 192,862 children born with congenital anomalies participated in the study. One registry was unable to get ethical approval to participate and linkage was not possible for two registries due to local reasons. Eleven registries linked to vital statistics and seven registries linked to mortality records only; one of the latter only had identification numbers for 78% of cases, hence it was excluded from further analysis. For registries linking to vital statistics: six linked over 95% of their cases for all years and five were unable to link at least 85% of all live born CA children in the earlier years of the study. No estimate of linkage success could be calculated for registries linking to mortality records. Irrespective of linkage method, deaths that occurred during the first week of life were over three times less likely to be linked compared to deaths occurring after the first week of life. Linkage to vital statistics can provide accurate estimates of survival of children with CAs in some European countries. Bias arises when linkage is not successful, as early neonatal deaths were less likely to be linked. Linkage to mortality records only cannot be recommended, as linkage quality, and hence bias, cannot be assessed.

Original language	English
Article number	e0256535
Number of pages	15
Journal	PLoS ONE
Volume	16
Issue number	8
Early online date	27 Aug 2021
DOIs	https://doi.org/10.1371/journal.pone.0256535
Publication status	Published online - 27 Aug 2021

Bibliographical note

Funding Information:
All authors (ML, JEG, JT, AR, DA, GA, IB, NB, LBB, CCC, OMC, AC, JD, ED, EG, MG, SVG, AH, EDH, SJ, BK, SKK, SK, KK, NL, LRL, AJN, LO, AP, AR, MS, IS, GT, DT, HEKdeW, DW, OZ, and JKM) were funded by the European Union's Horizon 2020 Research and Innovation programme. Grant agreement number 733001 (https://cordis.europa.eu/search?q=contenttype% 3D%27project%27%20AND%20(programme% 2Fcode%3D%27H2020-EU.3.1.%27)%20AND% 20(%27EUROLINKCAT%27)&p=1&num=10&srt= Relevance:decreasing). The funders had no role in study design, data collection and analysis, decision to publish, or preparation of the manuscript. We thank Prof Jennifer Kurinczuk, CAROBB, University of Oxford, and Dan Thayer, University of Swansea for their contribution to the project.

Publisher Copyright:
© 2021 Loane et al.This is an open access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited.

Keywords

Congenital anomalies
Survival
Linkage study

UN SDGs

This output contributes to the following UN Sustainable Development Goals (SDGs)

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10.1371/journal.pone.0256535Licence: CC BY

journal.pone.0256535 (2)Final published version, 1.05 MBLicence: CC BY

Cite this

Loane, M., Given, J., Tan, J., Reid, A., Akhmedzhanova, D., Astolfi, G., Barišić, I., Bertille, N., Bonet, LB., Cavero-Carbonell, C., Mokoroa Carollo, O., Coi, A., Densem, J., Draper, E., Garne, E., Gatt, M., Glinianaia, SV., Heino, A., Den Hond, E., ... Morris, JK. (2021). Linking a European cohort of children born with congenital anomalies to vital statistics and mortality records: a EUROlinkCAT study. PLoS ONE, 16(8), Article e0256535. Advance online publication. https://doi.org/10.1371/journal.pone.0256535

@article{917f7a2a87e4493699a0f16006166926,

title = "Linking a European cohort of children born with congenital anomalies to vital statistics and mortality records: a EUROlinkCAT study",

abstract = "EUROCAT is a European network of population-based congenital anomaly (CA)registries. Twenty-one registries agreed to participate in the EUROlinkCAT study to determine if reliable information on the survival of children born with a major CA between 1995 and 2014 can be obtained through linkage to national vital statistics or mortality records. Live birth children with a CA could be linked using personal identifiers to either their national vital statistics (including birth records, death records, hospital records) or to mortality records only, depending on the data available within each region. In total, 18 of 21 registries with data on 192,862 children born with congenital anomalies participated in the study. One registry was unable to get ethical approval to participate and linkage was not possible for two registries due to local reasons. Eleven registries linked to vital statistics and seven registries linked to mortality records only; one of the latter only had identification numbers for 78% of cases, hence it was excluded from further analysis. For registries linking to vital statistics: six linked over 95% of their cases for all years and five were unable to link at least 85% of all live born CA children in the earlier years of the study. No estimate of linkage success could be calculated for registries linking to mortality records. Irrespective of linkage method, deaths that occurred during the first week of life were over three times less likely to be linked compared to deaths occurring after the first week of life. Linkage to vital statistics can provide accurate estimates of survival of children with CAs in some European countries. Bias arises when linkage is not successful, as early neonatal deaths were less likely to be linked. Linkage to mortality records only cannot be recommended, as linkage quality, and hence bias, cannot be assessed.",

keywords = "Congenital anomalies, Survival, Linkage study",

author = "Maria Loane and Joanne Given and Joachim Tan and Abigail Reid and Diana Akhmedzhanova and Gianni Astolfi and Ingeborg Bari{\v s}i{\'c} and Nathalie Bertille and LB Bonet and Clara Cavero-Carbonell and {Mokoroa Carollo}, O and Alessio Coi and J Densem and E Draper and E Garne and M Gatt and SV Glinianaia and A Heino and {Den Hond}, E and S Jordan and B Khoshnood and S Kiuru-Kuhlefelt and K Klungsoyr and N Lelong and Lutke, {L. Ren{\'e}e} and AJ Neville and L Ostapchuk and A Puccini and A Rissmann and M Santoro and I Scanlon and G Thys and D Tucker and SK Urhoj and {de Walle}, HEK and D Wellesley and O Zurriaga and JK Morris",

note = "Funding Information: All authors (ML, JEG, JT, AR, DA, GA, IB, NB, LBB, CCC, OMC, AC, JD, ED, EG, MG, SVG, AH, EDH, SJ, BK, SKK, SK, KK, NL, LRL, AJN, LO, AP, AR, MS, IS, GT, DT, HEKdeW, DW, OZ, and JKM) were funded by the European Union's Horizon 2020 Research and Innovation programme. Grant agreement number 733001 (https://cordis.europa.eu/search?q=contenttype% 3D%27project%27%20AND%20(programme% 2Fcode%3D%27H2020-EU.3.1.%27)%20AND% 20(%27EUROLINKCAT%27)&p=1&num=10&srt= Relevance:decreasing). The funders had no role in study design, data collection and analysis, decision to publish, or preparation of the manuscript. We thank Prof Jennifer Kurinczuk, CAROBB, University of Oxford, and Dan Thayer, University of Swansea for their contribution to the project. Publisher Copyright: {\textcopyright} 2021 Loane et al.This is an open access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited.",

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doi = "10.1371/journal.pone.0256535",

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Loane, M , Given, J, Tan, J, Reid, A, Akhmedzhanova, D, Astolfi, G, Barišić, I, Bertille, N, Bonet, LB, Cavero-Carbonell, C, Mokoroa Carollo, O, Coi, A, Densem, J, Draper, E, Garne, E, Gatt, M, Glinianaia, SV, Heino, A, Den Hond, E, Jordan, S, Khoshnood, B, Kiuru-Kuhlefelt, S, Klungsoyr, K, Lelong, N, Lutke, LR, Neville, AJ, Ostapchuk, L, Puccini, A, Rissmann, A, Santoro, M, Scanlon, I, Thys, G, Tucker, D, Urhoj, SK, de Walle, HEK, Wellesley, D, Zurriaga, O & Morris, JK 2021, 'Linking a European cohort of children born with congenital anomalies to vital statistics and mortality records: a EUROlinkCAT study', PLoS ONE, vol. 16, no. 8, e0256535. https://doi.org/10.1371/journal.pone.0256535

TY - JOUR

T1 - Linking a European cohort of children born with congenital anomalies to vital statistics and mortality records: a EUROlinkCAT study

AU - Loane, Maria

AU - Given, Joanne

AU - Tan, Joachim

AU - Reid, Abigail

AU - Akhmedzhanova, Diana

AU - Astolfi, Gianni

AU - Barišić, Ingeborg

AU - Bertille, Nathalie

AU - Bonet, LB

AU - Cavero-Carbonell, Clara

AU - Mokoroa Carollo, O

AU - Coi, Alessio

AU - Densem, J

AU - Draper, E

AU - Garne, E

AU - Gatt, M

AU - Glinianaia, SV

AU - Heino, A

AU - Den Hond, E

AU - Jordan, S

AU - Khoshnood, B

AU - Kiuru-Kuhlefelt, S

AU - Klungsoyr, K

AU - Lelong, N

AU - Lutke, L. Renée

AU - Neville, AJ

AU - Ostapchuk, L

AU - Puccini, A

AU - Rissmann, A

AU - Santoro, M

AU - Scanlon, I

AU - Thys, G

AU - Tucker, D

AU - Urhoj, SK

AU - de Walle, HEK

AU - Wellesley, D

AU - Zurriaga, O

AU - Morris, JK

N1 - Funding Information: All authors (ML, JEG, JT, AR, DA, GA, IB, NB, LBB, CCC, OMC, AC, JD, ED, EG, MG, SVG, AH, EDH, SJ, BK, SKK, SK, KK, NL, LRL, AJN, LO, AP, AR, MS, IS, GT, DT, HEKdeW, DW, OZ, and JKM) were funded by the European Union's Horizon 2020 Research and Innovation programme. Grant agreement number 733001 (https://cordis.europa.eu/search?q=contenttype% 3D%27project%27%20AND%20(programme% 2Fcode%3D%27H2020-EU.3.1.%27)%20AND% 20(%27EUROLINKCAT%27)&p=1&num=10&srt= Relevance:decreasing). The funders had no role in study design, data collection and analysis, decision to publish, or preparation of the manuscript. We thank Prof Jennifer Kurinczuk, CAROBB, University of Oxford, and Dan Thayer, University of Swansea for their contribution to the project. Publisher Copyright: © 2021 Loane et al.This is an open access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited.

PY - 2021/8/27

Y1 - 2021/8/27

N2 - EUROCAT is a European network of population-based congenital anomaly (CA)registries. Twenty-one registries agreed to participate in the EUROlinkCAT study to determine if reliable information on the survival of children born with a major CA between 1995 and 2014 can be obtained through linkage to national vital statistics or mortality records. Live birth children with a CA could be linked using personal identifiers to either their national vital statistics (including birth records, death records, hospital records) or to mortality records only, depending on the data available within each region. In total, 18 of 21 registries with data on 192,862 children born with congenital anomalies participated in the study. One registry was unable to get ethical approval to participate and linkage was not possible for two registries due to local reasons. Eleven registries linked to vital statistics and seven registries linked to mortality records only; one of the latter only had identification numbers for 78% of cases, hence it was excluded from further analysis. For registries linking to vital statistics: six linked over 95% of their cases for all years and five were unable to link at least 85% of all live born CA children in the earlier years of the study. No estimate of linkage success could be calculated for registries linking to mortality records. Irrespective of linkage method, deaths that occurred during the first week of life were over three times less likely to be linked compared to deaths occurring after the first week of life. Linkage to vital statistics can provide accurate estimates of survival of children with CAs in some European countries. Bias arises when linkage is not successful, as early neonatal deaths were less likely to be linked. Linkage to mortality records only cannot be recommended, as linkage quality, and hence bias, cannot be assessed.

AB - EUROCAT is a European network of population-based congenital anomaly (CA)registries. Twenty-one registries agreed to participate in the EUROlinkCAT study to determine if reliable information on the survival of children born with a major CA between 1995 and 2014 can be obtained through linkage to national vital statistics or mortality records. Live birth children with a CA could be linked using personal identifiers to either their national vital statistics (including birth records, death records, hospital records) or to mortality records only, depending on the data available within each region. In total, 18 of 21 registries with data on 192,862 children born with congenital anomalies participated in the study. One registry was unable to get ethical approval to participate and linkage was not possible for two registries due to local reasons. Eleven registries linked to vital statistics and seven registries linked to mortality records only; one of the latter only had identification numbers for 78% of cases, hence it was excluded from further analysis. For registries linking to vital statistics: six linked over 95% of their cases for all years and five were unable to link at least 85% of all live born CA children in the earlier years of the study. No estimate of linkage success could be calculated for registries linking to mortality records. Irrespective of linkage method, deaths that occurred during the first week of life were over three times less likely to be linked compared to deaths occurring after the first week of life. Linkage to vital statistics can provide accurate estimates of survival of children with CAs in some European countries. Bias arises when linkage is not successful, as early neonatal deaths were less likely to be linked. Linkage to mortality records only cannot be recommended, as linkage quality, and hence bias, cannot be assessed.

KW - Congenital anomalies

KW - Survival

KW - Linkage study

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U2 - 10.1371/journal.pone.0256535

DO - 10.1371/journal.pone.0256535

M3 - Article

C2 - 34449798

SN - 1932-6203

VL - 16

JO - PLoS ONE

JF - PLoS ONE

IS - 8

M1 - e0256535

ER -

Linking a European cohort of children born with congenital anomalies to vital statistics and mortality records: a EUROlinkCAT study

Abstract

Bibliographical note

Keywords

UN SDGs

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

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