BMJ Open (Jun 2021)
EUROlinkCAT protocol for a European population-based data linkage study investigating the survival, morbidity and education of children with congenital anomalies
- ,
- Nathalie Lelong,
- Babak Khoshnood,
- David Tucker,
- Mika Gissler,
- Maria Loane,
- Ingeborg Barisic,
- Miriam Gatt,
- Kari Klungsoyr,
- Amanda Neville,
- Anna Pierini,
- Anke Rissmann,
- Oscar Zurriaga,
- Joan K Morris,
- Paula Braz,
- Vera Nelen,
- Judith Rankin,
- Ester Garne,
- Sue Jordan,
- Anna Heino,
- Silvia Baldacci,
- Michele Santoro,
- Alessio Coi,
- Lorena Mezzasalma,
- Daniel Thayer,
- Svetlana V Glinianaia,
- Hermien de Walle,
- Guy Thys,
- Anna Latos-Bielenska,
- Aurora Puccini,
- Elisa Ballardini,
- Wladimir Wertelecki,
- Carlos Matias Dias,
- Gianni Astolfi,
- James Densem,
- Joachim Tan,
- Joanne Emma Given,
- Hugh Claridge,
- Abigail Reid,
- Liz Limb,
- Gillian Briggs,
- Nicholas Connor,
- Stine Kjaer Urhoj,
- Mads Damkjær,
- Christina Neergaard Pedersen,
- Annarita Armaroli,
- Ljubica Boban,
- L Renée Lutke,
- Nicole H A Siemensma-Mühlenberg,
- Nathalie Bertille,
- Makan Rahshenas,
- Clara Cavero Carbonell,
- Laia Barrachina Bonet,
- Laura García Villodre,
- Anna Jamry-Dziurla,
- Sonja Kiuru-Kuhlefelt,
- Lyubov Yevtushok,
- Nataliia Zymak-Zakutnia,
- Diana Akhmedzhanova,
- Lyubov Ostapchuk,
- Oksana Tsizh,
- Serhii Lapchenko,
- Ausenda Machado,
- Ana João Santos,
- Liliana Antunes,
- Hanitra Randrianaivo-Ranjatoelina,
- Elly Den Hond,
- Olatz Mokoroa Carollo,
- Ieuan Scanlon
Affiliations
- 3University College London
- Nathalie Lelong
- Babak Khoshnood
- Obstetrical, Perinatal and Pediatric Epidemiology Research Team (EPOPé), Center of Research in Epidemiology and Statistics (CRESS), Institut National de la Santé et de la Recherche Médicale (INSERM), INRA, Université de Paris, Paris, France
- David Tucker
- Public Health Wales National Health Service Trust, Cardiff, UK
- Mika Gissler
- Knowledge Brokers, Finnish Institute for Health and Welfare, Helsinki, Finland
- Maria Loane
- Institute of Nursing and Health Research, Faculty of Life and Health Sciences,Ulster University, Belfast, UK
- Ingeborg Barisic
- Children`s Hospital Zagreb, Centre of Excellence for Reproductive and Regenerative Medicine, Medical School University of Zagreb, Zagreb, Croatia
- Miriam Gatt
- Malta Congenital Anomalies Registry, Directorate for Health Information and Research, Pieta, Malta
- Kari Klungsoyr
- Department of Global Public Health and Primary Care, University of Bergen, Bergen, Norway
- Amanda Neville
- 10 Emilia Romagna Registry of Birth Defects and Center for Clinical and Epidemiological Research, University of Ferrara, Ferrara, Italy
- Anna Pierini
- Institute of Clinical Physiology National Research Council, Pisa, Italy
- Anke Rissmann
- Malformation Monitoring Centre Saxony-Anhalt, Medical Faculty, Otto-von-Guericke University, Magdeburg, Germany
- Oscar Zurriaga
- Rare Diseases Research Join Unit, Foundation for the Promotion of Health and Biomedical Research and Universitat de Valencia, Valencia, Spain
- Joan K Morris
- Population Health Research Institute, St George`s University of London, London, UK
- Paula Braz
- Vera Nelen
- Provincial Institute for Hygiene, Antwerp, Belgium
- Judith Rankin
- Population Health Sciences Institute, Newcastle University, Newcastle upon Tyne, UK
- Ester Garne
- Department of Paediatrics and Adolescent Medicine, Lillebaelt Hospital, University Hospital of Southern Denmark, Kolding, Denmark
- Sue Jordan
- Faculty of Medicine, Health and Life Science, Swansea University, Swansea, UK
- Anna Heino
- Department of Knowledge Brokers, THL Finnish Institute for Health and Welfare, Helsinki, Finland
- Silvia Baldacci
- 3 Unit of Epidemiology of Rare Diseases and Congenital Anomalies, Institute of Clinical Physiology, National Research Council, Pisa, Italy
- Michele Santoro
- Unit of Epidemiology of Rare Diseases and Congenital Anomalies, Institute of Clinical Physiology National Research Council, Pisa, Italy
- Alessio Coi
- Institute of Clinical Physiology, National Research Council Pisa Research Area, Pisa, Italy
- Lorena Mezzasalma
- Daniel Thayer
- Svetlana V Glinianaia
- Population Health Sciences Institute, Newcastle University, Newcastle upon Tyne, UK
- Hermien de Walle
- Department of Genetics, University Medical Center Groningen, University of Groningen, Groningen, The Netherlands
- Guy Thys
- Anna Latos-Bielenska
- Chair and Department of Medical Genetics, University of Medical Sciences, Poznan, Poland
- Aurora Puccini
- Drug and Medical Device Area, Emilia Romagna Health Department, Emilia-Romagna Regional Healthcare Services, Bologna, Emilia-Romagna, Italy
- Elisa Ballardini
- Department of Neuroscience and Rehabilitation, University of Ferrara, Ferrara, Italy
- Wladimir Wertelecki
- OMNI-Net for Children International Charitable Fund, Rivne, Ukraine
- Carlos Matias Dias
- Gianni Astolfi
- Emilia Romagna Registry of Birth Defects, University of Ferrara, Ferrara, Italy
- James Densem
- Biomedical Computing Limited, Battle, UK
- Joachim Tan
- Population Health Research Institute, St George`s University of London, London, UK
- Joanne Emma Given
- Institute of Nursing and Health Research, Faculty of Life and Health Sciences,Ulster University, Belfast, UK
- Hugh Claridge
- School of Health Sciences, Faculty of Health and Medical Sciences, University of Surrey, Guildford, UK
- Abigail Reid
- Population Health Research Institute, St George`s University of London, London, UK
- Liz Limb
- Gillian Briggs
- Population Health Research Institute, St George`s University of London, London, UK
- Nicholas Connor
- Stine Kjaer Urhoj
- associate professor
- Mads Damkjær
- Department of Paediatrics and Adolescent Medicine, Lillebaelt Hospital - University Hospital of Southern Denmark, Kolding, Denmark
- Christina Neergaard Pedersen
- Annarita Armaroli
- Ljubica Boban
- L Renée Lutke
- Nicole H A Siemensma-Mühlenberg
- Nathalie Bertille
- Makan Rahshenas
- Clara Cavero Carbonell
- Laia Barrachina Bonet
- Laura García Villodre
- Anna Jamry-Dziurla
- Chair and Department of Medical Genetics, University of Medical Sciences, Poznan, Poland
- Sonja Kiuru-Kuhlefelt
- Knowledge Brokers, Finnish Institute for Health and Welfare, Helsinki, Finland
- Lyubov Yevtushok
- OMNI-Net for Children International Charitable Fund, Rivne Regional Medical Diagnostic Center, Rivne, Ukraine
- Nataliia Zymak-Zakutnia
- Diana Akhmedzhanova
- OMNI-Net for Children International Charitable Fund, Rivne Regional Medical Diagnostic Center, Rivne, Ukraine
- Lyubov Ostapchuk
- Oksana Tsizh
- Serhii Lapchenko
- Ausenda Machado
- Department of Epidemiology, National Institute of Health Doutor Ricardo Jorge, Lisbon, Portugal
- Ana João Santos
- Department of Epidemiology, National Institute of Health Doutor Ricardo Jorge, Lisbon, Portugal
- Liliana Antunes
- Hanitra Randrianaivo-Ranjatoelina
- Elly Den Hond
- Olatz Mokoroa Carollo
- Ieuan Scanlon
- Department of Nursing, Swansea University, Swansea, UK
- DOI
- https://doi.org/10.1136/bmjopen-2020-047859
- Journal volume & issue
-
Vol. 11,
no. 6
Abstract
Introduction Congenital anomalies (CAs) are a major cause of infant mortality, childhood morbidity and long-term disability. Over 130 000 children born in Europe every year will have a CA. This paper describes the EUROlinkCAT study, which is investigating the health and educational outcomes of children with CAs for the first 10 years of their lives.Methods and analysis EUROCAT is a European network of population-based registries for the epidemiological surveillance of CAs. EUROlinkCAT is using the EUROCAT infrastructure to support 22 EUROCAT registries in 14 countries to link their data on births with CAs to mortality, hospital discharge, prescription and educational databases. Once linked, each registry transforms their case data into a common data model (CDM) format and they are then supplied with common STATA syntax scripts to analyse their data. The resulting aggregate tables and analysis results are submitted to a central results repository (CRR) and meta-analyses are performed to summarise the results across all registries. The CRR currently contains data on 155 594 children with a CA followed up to age 10 from a population of 6 million births from 1995 to 2014.Ethics The CA registries have the required ethics permissions for routine surveillance and transmission of anonymised data to the EUROCAT central database. Each registry is responsible for applying for and obtaining additional ethics and other permissions required for their participation in EUROlinkCAT.Dissemination The CDM and associated documentation, including linkage and standardisation procedures, will be available post-EUROlinkCAT thus facilitating future local, national and European-level analyses to improve healthcare. Recommendations to improve the accuracy of routinely collected data will be made.Findings will provide evidence to inform parents, health professionals, public health authorities and national treatment guidelines to optimise diagnosis, prevention and treatment for these children with a view to reducing health inequalities in Europe.
