Periodic Reporting for period 4 - EUROlinkCAT (EUROlinkCAT: Establishing a linked European Cohort of Children with Congenital Anomalies)
Periodo di rendicontazione: 2021-07-01 al 2022-05-31
Issue being addressed
- Over 130000 children born in Europe every year will have a major congenital anomaly (CA)
- CAs (birth defects) are a major cause of infant mortality, childhood morbidity and long-term disability
- There is a lack of evidence-based information on the survival, health and educational achievements of children born with CAs in Europe
Importance to society
- Investigating the health and educational outcomes of children with CAs over their first 10 years of life will allow parents, health professionals, public health and education authorities to plan for, and make informed decisions about, the treatment, services and education arrangements for these children. This will result in improved care, support and quality of life of these children.
- The project will help to facilitate the development of a reciprocal relationship between families with children with CAs, health and social care professionals and researchers via a multi-national survey and focus groups. These aim to provide the opportunity for public and professional engagement in setting and disseminating relevant research priorities and their outcomes.
Objectives
- Establish a European network of standardised datasets containing information on the mortality, health, educational achievements and needs of children with CAs born 1995-2014 up until 10 years of age and enable these data to be available for European analyses beyond the end of the project.
- Expand knowledge on the survival, health, disease determinants, educational achievements and needs of children according to their specific CA and risk factors including socio-economic health inequalities
- Evaluate the costs of hospitalisation during the first five years of life for children with a CA
- Evaluate the accuracy of existing electronic hospital databases and make recommendations to improve their use
- Consider the experiences of parents of children with spina bifida, cleft lip, Down syndrome or needing heart surgery to ensure research findings are relevant
- Engage with international/national/regional health authorities to ensure that findings are implemented and translated into health policy
- Over 130000 children born in Europe every year will have a major congenital anomaly (CA)
- CAs (birth defects) are a major cause of infant mortality, childhood morbidity and long-term disability
- There is a lack of evidence-based information on the survival, health and educational achievements of children born with CAs in Europe
Importance to society
- Investigating the health and educational outcomes of children with CAs over their first 10 years of life will allow parents, health professionals, public health and education authorities to plan for, and make informed decisions about, the treatment, services and education arrangements for these children. This will result in improved care, support and quality of life of these children.
- The project will help to facilitate the development of a reciprocal relationship between families with children with CAs, health and social care professionals and researchers via a multi-national survey and focus groups. These aim to provide the opportunity for public and professional engagement in setting and disseminating relevant research priorities and their outcomes.
Objectives
- Establish a European network of standardised datasets containing information on the mortality, health, educational achievements and needs of children with CAs born 1995-2014 up until 10 years of age and enable these data to be available for European analyses beyond the end of the project.
- Expand knowledge on the survival, health, disease determinants, educational achievements and needs of children according to their specific CA and risk factors including socio-economic health inequalities
- Evaluate the costs of hospitalisation during the first five years of life for children with a CA
- Evaluate the accuracy of existing electronic hospital databases and make recommendations to improve their use
- Consider the experiences of parents of children with spina bifida, cleft lip, Down syndrome or needing heart surgery to ensure research findings are relevant
- Engage with international/national/regional health authorities to ensure that findings are implemented and translated into health policy
Overview
EUROlinkCAT formed a consortium of 22 EUROCAT registries in 14 European countries who, where possible, linked children with a CA born 1995-2014 up to age 10 to mortality, hospital discharge, prescription and education databases. Each registry standardised their data and ran centrally provided syntax scripts to create standard summarised tables and analytic results. These results were submitted to a Central Results Repository (CRR), enabling hypotheses on their health and education to be investigated at an EU level by researchers. Focus groups and a multi-national survey involved parents of children with CAs in setting research priorities and provided data on how they wished to obtain information about their children’s health conditions.
European network of standardised datasets
Ethics permissions were obtained and linkage and standardisation were performed for 17 registries with mortality data, 12 with health data, 7 with prescription data, 6 with education data and 3 with data on Terminations of Pregnancies for Fetal Anomalies (TOPFAs). 180,000 children with a CA and 2,000,000 without a CA were linked. Information on the ethics required, the variables available in the data sources, the methods of linkage and standardisation and the corresponding EUROlinkCAT common data models is available in published papers and publicly available reports.
EUROlinkCAT Central Results Repository (CRR)
Registries ran Stata analysis scripts and the analytic results relating to mortality, morbidity and education have been deposited in the CRR. A manual describing the contents of the CRR is available. A data request form for the results in the CRR is available on the EUROlinkCAT website and interactive website graphs can be created.
Main Results
Mortality
Ten-year survival estimates are available for children with 72 different CAs - survival improved by over 30% for those born 2005-2014 compared to 1995-2004.
Morbidity
During their first year of life, 85% of children with a CA were admitted to hospital compared to 31% of children without a CA, and once admitted these children spent 2-3 times longer in hospital. Almost 40% of children with a CA had surgery within their first year of life, compared to under 1% of children without a CA. This information is also available for 83 different CAs for children up to 10 years of age. Almost half of all children with severe congenital heart defects were prescribed a cardiovascular medication in their first year of life, with one fifth having prescriptions up to age 5. Information on anti-diabetic medications, anti-convulsants, anti-asthmatics and antibiotics is also available for 33 different CAs.
Educational achievements
Linking health and education data was achieved in only 4 countries due a lack of national education databases and concern over linking health data to other data. The majority of children with a CA did achieve the expected academic levels by age 16, although they had more special education support up to age 11 years. A lower proportion of them applied to continue further education, compared to children without a CA.
Use of electronic hospital databases
Data from CA registries are needed for the accurate monitoring and research of CAs, as only two-thirds of live born children with a CA had their CA diagnosis code recorded correctly in electronic hospital databases. A computer algorithm to identify hospital records of children whose clinical notes need to be examined by a clinician to determine if they have a CA, was refined to enable data in CA registries to be enriched. This was one of the recommendations in the report on guidelines for improving the quality of the congenital anomaly coding.
Experience of Parents of Children with CA
The wish to have positive messages about their child’s potential was the main message from the parents in several face-to-face focus groups and from a multi-lingual internet survey of 1,109 people, covering 10 countries in Europe disseminated using social media and parent support group contacts.
Dissemination
Dissemination up to 31/05/2022 has included the conference in Poznań, Poland with 720 registered to attend from 59 countries, 12 papers accepted by or published in peer reviewed journals (with 6 more under review and 21 drafted), as well as a targeted report to EU institutions hosting health care databases. Parents of children with a CA have been involved throughout and were active on social media via EUROlinkCAT’s twitter account. A dedicated “Parents” area on the website and lay summaries and infographics in 10 languages are available.
EUROlinkCAT formed a consortium of 22 EUROCAT registries in 14 European countries who, where possible, linked children with a CA born 1995-2014 up to age 10 to mortality, hospital discharge, prescription and education databases. Each registry standardised their data and ran centrally provided syntax scripts to create standard summarised tables and analytic results. These results were submitted to a Central Results Repository (CRR), enabling hypotheses on their health and education to be investigated at an EU level by researchers. Focus groups and a multi-national survey involved parents of children with CAs in setting research priorities and provided data on how they wished to obtain information about their children’s health conditions.
European network of standardised datasets
Ethics permissions were obtained and linkage and standardisation were performed for 17 registries with mortality data, 12 with health data, 7 with prescription data, 6 with education data and 3 with data on Terminations of Pregnancies for Fetal Anomalies (TOPFAs). 180,000 children with a CA and 2,000,000 without a CA were linked. Information on the ethics required, the variables available in the data sources, the methods of linkage and standardisation and the corresponding EUROlinkCAT common data models is available in published papers and publicly available reports.
EUROlinkCAT Central Results Repository (CRR)
Registries ran Stata analysis scripts and the analytic results relating to mortality, morbidity and education have been deposited in the CRR. A manual describing the contents of the CRR is available. A data request form for the results in the CRR is available on the EUROlinkCAT website and interactive website graphs can be created.
Main Results
Mortality
Ten-year survival estimates are available for children with 72 different CAs - survival improved by over 30% for those born 2005-2014 compared to 1995-2004.
Morbidity
During their first year of life, 85% of children with a CA were admitted to hospital compared to 31% of children without a CA, and once admitted these children spent 2-3 times longer in hospital. Almost 40% of children with a CA had surgery within their first year of life, compared to under 1% of children without a CA. This information is also available for 83 different CAs for children up to 10 years of age. Almost half of all children with severe congenital heart defects were prescribed a cardiovascular medication in their first year of life, with one fifth having prescriptions up to age 5. Information on anti-diabetic medications, anti-convulsants, anti-asthmatics and antibiotics is also available for 33 different CAs.
Educational achievements
Linking health and education data was achieved in only 4 countries due a lack of national education databases and concern over linking health data to other data. The majority of children with a CA did achieve the expected academic levels by age 16, although they had more special education support up to age 11 years. A lower proportion of them applied to continue further education, compared to children without a CA.
Use of electronic hospital databases
Data from CA registries are needed for the accurate monitoring and research of CAs, as only two-thirds of live born children with a CA had their CA diagnosis code recorded correctly in electronic hospital databases. A computer algorithm to identify hospital records of children whose clinical notes need to be examined by a clinician to determine if they have a CA, was refined to enable data in CA registries to be enriched. This was one of the recommendations in the report on guidelines for improving the quality of the congenital anomaly coding.
Experience of Parents of Children with CA
The wish to have positive messages about their child’s potential was the main message from the parents in several face-to-face focus groups and from a multi-lingual internet survey of 1,109 people, covering 10 countries in Europe disseminated using social media and parent support group contacts.
Dissemination
Dissemination up to 31/05/2022 has included the conference in Poznań, Poland with 720 registered to attend from 59 countries, 12 papers accepted by or published in peer reviewed journals (with 6 more under review and 21 drafted), as well as a targeted report to EU institutions hosting health care databases. Parents of children with a CA have been involved throughout and were active on social media via EUROlinkCAT’s twitter account. A dedicated “Parents” area on the website and lay summaries and infographics in 10 languages are available.
This project has been the first to establish a comprehensive set of independent standardised datasets containing uniformly coded and verified information on the morbidity and mortality of children with CAs up to age 10 across Europe. For many CAs these are the first published European survival estimates and morbidity outcomes up to 10 years of age and education results up to 16 years of age.. The standardised datasets and the CRR are invaluable resources for research on improving the survival and morbidity of children with CAs. EUROlinkCAT has established methods to enable routine electronic data to be used for both research and surveillance of CAs across Europe.