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

Anke Rissmann Orcid Logo, Joachim Tan Orcid Logo, Svetlana V Glinianaia, Judith Rankin, Anna Pierini, Michele Santoro, Alessio Coi, Ester Garne, Maria Loane, Joanne Given, Abigail Reid, Amaia Aizpurua, Diana Akhmedzhanova, Elisa Ballardini, Ingeborg Barisic Orcid Logo, Clara Cavero-Carbonell Orcid Logo, Hermien E K de Walle, Miriam Gatt, Mika Gissler, Anna Heino, Sue Jordan Orcid Logo, Stine Kjaer Urhoj Orcid Logo, Kari Klungsøyr, Renee Lutke, Olatz Mokoroa, Amanda Julie Neville, Daniel S Thayer, Diana G Wellesley, Lyubov Yevtushok, Oscar Zurriaga, Joan Morris Orcid Logo

BMJ Paediatrics Open, Volume: 7, Issue: 1, Start page: e001617

Swansea University Author: Sue Jordan Orcid Logo

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Abstract

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 Januar...

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Published in: BMJ Paediatrics Open
ISSN: 2399-9772
Published: BMJ 2023
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fullrecord <?xml version="1.0" encoding="utf-8"?><rfc1807 xmlns:xsi="http://www.w3.org/2001/XMLSchema-instance" xmlns:xsd="http://www.w3.org/2001/XMLSchema"><bib-version>v2</bib-version><id>64004</id><entry>2023-07-31</entry><title>Causes of death in children with congenital anomalies up to age 10 in eight European countries</title><swanseaauthors><author><sid>24ce9db29b4bde1af4e83b388aae0ea1</sid><ORCID>0000-0002-5691-2987</ORCID><firstname>Sue</firstname><surname>Jordan</surname><name>Sue Jordan</name><active>true</active><ethesisStudent>false</ethesisStudent></author></swanseaauthors><date>2023-07-31</date><deptcode>HNU</deptcode><abstract>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.</abstract><type>Journal Article</type><journal>BMJ Paediatrics Open</journal><volume>7</volume><journalNumber>1</journalNumber><paginationStart>e001617</paginationStart><paginationEnd/><publisher>BMJ</publisher><placeOfPublication/><isbnPrint/><isbnElectronic/><issnPrint/><issnElectronic>2399-9772</issnElectronic><keywords>Infant and child mortality, death, congenital anomalies</keywords><publishedDay>30</publishedDay><publishedMonth>6</publishedMonth><publishedYear>2023</publishedYear><publishedDate>2023-06-30</publishedDate><doi>10.1136/bmjpo-2022-001617</doi><url>http://dx.doi.org/10.1136/bmjpo-2022-001617</url><notes>Data are available on reasonable request. The aggregate data that support the findings of this study are available from the authors for scientifically valid requests and with permission of the participating registries of congenital anomalies.</notes><college>COLLEGE NANME</college><department>Nursing</department><CollegeCode>COLLEGE CODE</CollegeCode><DepartmentCode>HNU</DepartmentCode><institution>Swansea University</institution><apcterm>Another institution paid the OA fee</apcterm><funders>Horizon 2020</funders><projectreference>ELC</projectreference><lastEdited>2023-08-18T09:58:49.1697764</lastEdited><Created>2023-07-31T19:24:10.2844819</Created><path><level id="1">Faculty of Medicine, Health and Life Sciences</level><level id="2">Swansea University Medical School - Medicine</level></path><authors><author><firstname>Anke</firstname><surname>Rissmann</surname><orcid>0000-0002-9437-2790</orcid><order>1</order></author><author><firstname>Joachim</firstname><surname>Tan</surname><orcid>0000-0003-0462-4761</orcid><order>2</order></author><author><firstname>Svetlana V</firstname><surname>Glinianaia</surname><order>3</order></author><author><firstname>Judith</firstname><surname>Rankin</surname><order>4</order></author><author><firstname>Anna</firstname><surname>Pierini</surname><order>5</order></author><author><firstname>Michele</firstname><surname>Santoro</surname><order>6</order></author><author><firstname>Alessio</firstname><surname>Coi</surname><order>7</order></author><author><firstname>Ester</firstname><surname>Garne</surname><order>8</order></author><author><firstname>Maria</firstname><surname>Loane</surname><order>9</order></author><author><firstname>Joanne</firstname><surname>Given</surname><order>10</order></author><author><firstname>Abigail</firstname><surname>Reid</surname><order>11</order></author><author><firstname>Amaia</firstname><surname>Aizpurua</surname><order>12</order></author><author><firstname>Diana</firstname><surname>Akhmedzhanova</surname><order>13</order></author><author><firstname>Elisa</firstname><surname>Ballardini</surname><order>14</order></author><author><firstname>Ingeborg</firstname><surname>Barisic</surname><orcid>0000-0002-9085-6747</orcid><order>15</order></author><author><firstname>Clara</firstname><surname>Cavero-Carbonell</surname><orcid>0000-0002-4858-6456</orcid><order>16</order></author><author><firstname>Hermien E K de</firstname><surname>Walle</surname><order>17</order></author><author><firstname>Miriam</firstname><surname>Gatt</surname><order>18</order></author><author><firstname>Mika</firstname><surname>Gissler</surname><order>19</order></author><author><firstname>Anna</firstname><surname>Heino</surname><order>20</order></author><author><firstname>Sue</firstname><surname>Jordan</surname><orcid>0000-0002-5691-2987</orcid><order>21</order></author><author><firstname>Stine Kjaer</firstname><surname>Urhoj</surname><orcid>0000-0002-2069-9723</orcid><order>22</order></author><author><firstname>Kari</firstname><surname>Klungsøyr</surname><order>23</order></author><author><firstname>Renee</firstname><surname>Lutke</surname><order>24</order></author><author><firstname>Olatz</firstname><surname>Mokoroa</surname><order>25</order></author><author><firstname>Amanda Julie</firstname><surname>Neville</surname><order>26</order></author><author><firstname>Daniel S</firstname><surname>Thayer</surname><order>27</order></author><author><firstname>Diana G</firstname><surname>Wellesley</surname><order>28</order></author><author><firstname>Lyubov</firstname><surname>Yevtushok</surname><order>29</order></author><author><firstname>Oscar</firstname><surname>Zurriaga</surname><order>30</order></author><author><firstname>Joan</firstname><surname>Morris</surname><orcid>0000-0002-7164-612x</orcid><order>31</order></author></authors><documents><document><filename>64004__28310__51d4c3e8d4214f5bb60790ab2c467d26.pdf</filename><originalFilename>64004.VOR.pdf</originalFilename><uploaded>2023-08-18T09:56:59.7545659</uploaded><type>Output</type><contentLength>1632447</contentLength><contentType>application/pdf</contentType><version>Version of Record</version><cronfaStatus>true</cronfaStatus><documentNotes>© 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. http://creativecommons.org/licenses/by-nc/4.0/ This is an open access article distributed in accordance with the Creative Commons Attribution Non Commercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited, appropriate credit is given, any changes made indicated, and the use is non-commercial.</documentNotes><copyrightCorrect>true</copyrightCorrect><language>eng</language><licence>http://creativecommons.org/licenses/by-nc/4.0/</licence></document></documents><OutputDurs/></rfc1807>
spelling v2 64004 2023-07-31 Causes of death in children with congenital anomalies up to age 10 in eight European countries 24ce9db29b4bde1af4e83b388aae0ea1 0000-0002-5691-2987 Sue Jordan Sue Jordan true false 2023-07-31 HNU 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. Journal Article BMJ Paediatrics Open 7 1 e001617 BMJ 2399-9772 Infant and child mortality, death, congenital anomalies 30 6 2023 2023-06-30 10.1136/bmjpo-2022-001617 http://dx.doi.org/10.1136/bmjpo-2022-001617 Data are available on reasonable request. The aggregate data that support the findings of this study are available from the authors for scientifically valid requests and with permission of the participating registries of congenital anomalies. COLLEGE NANME Nursing COLLEGE CODE HNU Swansea University Another institution paid the OA fee Horizon 2020 ELC 2023-08-18T09:58:49.1697764 2023-07-31T19:24:10.2844819 Faculty of Medicine, Health and Life Sciences Swansea University Medical School - Medicine Anke Rissmann 0000-0002-9437-2790 1 Joachim Tan 0000-0003-0462-4761 2 Svetlana V Glinianaia 3 Judith Rankin 4 Anna Pierini 5 Michele Santoro 6 Alessio Coi 7 Ester Garne 8 Maria Loane 9 Joanne Given 10 Abigail Reid 11 Amaia Aizpurua 12 Diana Akhmedzhanova 13 Elisa Ballardini 14 Ingeborg Barisic 0000-0002-9085-6747 15 Clara Cavero-Carbonell 0000-0002-4858-6456 16 Hermien E K de Walle 17 Miriam Gatt 18 Mika Gissler 19 Anna Heino 20 Sue Jordan 0000-0002-5691-2987 21 Stine Kjaer Urhoj 0000-0002-2069-9723 22 Kari Klungsøyr 23 Renee Lutke 24 Olatz Mokoroa 25 Amanda Julie Neville 26 Daniel S Thayer 27 Diana G Wellesley 28 Lyubov Yevtushok 29 Oscar Zurriaga 30 Joan Morris 0000-0002-7164-612x 31 64004__28310__51d4c3e8d4214f5bb60790ab2c467d26.pdf 64004.VOR.pdf 2023-08-18T09:56:59.7545659 Output 1632447 application/pdf Version of Record true © 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. http://creativecommons.org/licenses/by-nc/4.0/ This is an open access article distributed in accordance with the Creative Commons Attribution Non Commercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited, appropriate credit is given, any changes made indicated, and the use is non-commercial. true eng http://creativecommons.org/licenses/by-nc/4.0/
title Causes of death in children with congenital anomalies up to age 10 in eight European countries
spellingShingle Causes of death in children with congenital anomalies up to age 10 in eight European countries
Sue Jordan
title_short Causes of death in children with congenital anomalies up to age 10 in eight European countries
title_full Causes of death in children with congenital anomalies up to age 10 in eight European countries
title_fullStr Causes of death in children with congenital anomalies up to age 10 in eight European countries
title_full_unstemmed Causes of death in children with congenital anomalies up to age 10 in eight European countries
title_sort Causes of death in children with congenital anomalies up to age 10 in eight European countries
author_id_str_mv 24ce9db29b4bde1af4e83b388aae0ea1
author_id_fullname_str_mv 24ce9db29b4bde1af4e83b388aae0ea1_***_Sue Jordan
author Sue Jordan
author2 Anke Rissmann
Joachim Tan
Svetlana V Glinianaia
Judith Rankin
Anna Pierini
Michele Santoro
Alessio Coi
Ester Garne
Maria Loane
Joanne Given
Abigail Reid
Amaia Aizpurua
Diana Akhmedzhanova
Elisa Ballardini
Ingeborg Barisic
Clara Cavero-Carbonell
Hermien E K de Walle
Miriam Gatt
Mika Gissler
Anna Heino
Sue Jordan
Stine Kjaer Urhoj
Kari Klungsøyr
Renee Lutke
Olatz Mokoroa
Amanda Julie Neville
Daniel S Thayer
Diana G Wellesley
Lyubov Yevtushok
Oscar Zurriaga
Joan Morris
format Journal article
container_title BMJ Paediatrics Open
container_volume 7
container_issue 1
container_start_page e001617
publishDate 2023
institution Swansea University
issn 2399-9772
doi_str_mv 10.1136/bmjpo-2022-001617
publisher BMJ
college_str Faculty of Medicine, Health and Life Sciences
hierarchytype
hierarchy_top_id facultyofmedicinehealthandlifesciences
hierarchy_top_title Faculty of Medicine, Health and Life Sciences
hierarchy_parent_id facultyofmedicinehealthandlifesciences
hierarchy_parent_title Faculty of Medicine, Health and Life Sciences
department_str Swansea University Medical School - Medicine{{{_:::_}}}Faculty of Medicine, Health and Life Sciences{{{_:::_}}}Swansea University Medical School - Medicine
url http://dx.doi.org/10.1136/bmjpo-2022-001617
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description 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.
published_date 2023-06-30T09:58:50Z
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