{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,2,28]],"date-time":"2026-02-28T02:01:33Z","timestamp":1772244093711,"version":"3.50.1"},"reference-count":41,"publisher":"National Institute for Health and Care Research","license":[{"start":{"date-parts":[[2024,11,6]],"date-time":"2024-11-06T00:00:00Z","timestamp":1730851200000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"DOI":"10.13039\/501100007602","name":"Programme Grants for Applied Research","doi-asserted-by":"publisher","award":["NIHR202025"],"award-info":[{"award-number":["NIHR202025"]}],"id":[{"id":"10.13039\/501100007602","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100014013","name":"UK Research and Innovation","doi-asserted-by":"publisher","award":["ES\/V000977\/1;ES\/X003663\/1;ES\/X000427\/1"],"award-info":[{"award-number":["ES\/V000977\/1;ES\/X003663\/1;ES\/X000427\/1"]}],"id":[{"id":"10.13039\/100014013","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["openresearch.nihr.ac.uk"],"crossmark-restriction":false},"short-container-title":["NIHR Open Res"],"abstract":"<ns3:p>Background Major congenital anomalies (CAs) affect around 2% of live births and are a primary cause of infant mortality, childhood morbidity and long-term disability, often requiring hospitalisation and\/or surgery. Children with CAs are at greater risk of lower educational attainment compared with their peers, which could be due to learning disabilities, higher rates of ill-health and school absences, or lack of adequate educational support. Our study will compare the educational attainment of children with CAs to those of their peers up to age 11 in England, using linked administrative health and education data. Methods We will analyse data from the ECHILD (Education and Child Health Insights from Linked Data) database. Children born in NHS-funded hospitals from 1st September 2003 to 31st August 2008 whose hospital records were linked to their educational records at three Key Stages (ages 4\/5, 6\/7 and 10\/11 years) will be included. Children with different CAs, indicated by recorded hospital diagnosis codes, will be compared to children without CAs. We will compare the proportions of enrolled children who take the assessment, the proportions who reached national expected levels of attainment, and the mean standardised attainment scores for Maths and English at each Key Stage. We will describe variations in outcome by sex, ethnic minority background, region, and neighbourhood deprivation, and perform regression modelling to compare the attainment trajectories of children with and without CAs, controlling for sociodemographic factors. Ethics and dissemination Ethics approval has been obtained for the analyses of the ECHILD database. Our findings will provide information for parents regarding their children\u2019s expected academic potential, and also enable the development of interventions to support those at risk of not doing well. We will disseminate our findings to academics, policy makers, service users and providers through seminars, peer-reviewed publications, conference abstracts and other media (lay summaries and infographics).<\/ns3:p>","DOI":"10.3310\/nihropenres.13750.1","type":"journal-article","created":{"date-parts":[[2024,11,6]],"date-time":"2024-11-06T11:05:09Z","timestamp":1730891109000},"page":"68","update-policy":"https:\/\/doi.org\/10.3310\/nihropenres.crossmark-policy","source":"Crossref","is-referenced-by-count":1,"title":["Educational outcomes of children with major congenital anomalies: Study protocol for a population-based cohort study using linked hospital and education data from England"],"prefix":"10.3310","volume":"4","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-0462-4761","authenticated-orcid":false,"given":"Joachim","family":"Tan","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0009-0001-4034-4153","authenticated-orcid":false,"given":"Ayana","family":"Cant","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-1148-1017","authenticated-orcid":false,"given":"Kate","family":"Lewis","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-9776-6242","authenticated-orcid":false,"given":"Vincent","family":"Nguyen","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-1035-1448","authenticated-orcid":false,"given":"Ania","family":"Zylbersztejn","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-5456-1936","authenticated-orcid":false,"given":"Laura","family":"Gimeno","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Pia","family":"Hardelid","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-7853-0528","authenticated-orcid":false,"given":"Bianca","family":"De 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Wilkinson","year":"2017","journal-title":"J Pediatr Urol."},{"key":"ref-34","article-title":"Early years foundation stage profile: 2024 handbook","year":"2023"},{"key":"ref-35","article-title":"Primary school accountability in 2018: A technical guide for primary maintained schools, academies and free schools"},{"key":"ref-36","doi-asserted-by":"publisher","first-page":"1332-1360","DOI":"10.1037\/a0037173","article-title":"The Flynn effect: a meta-analysis.","volume":"140","author":"L Trahan","year":"2014","journal-title":"Psychol Bull."},{"key":"ref-37","doi-asserted-by":"publisher","first-page":"563-568","DOI":"10.1136\/archdischild-2023-325310","article-title":"Longitudinal educational attainment among children with isolated oral cleft: a cohort study.","volume":"108","author":"M Park","year":"2023","journal-title":"Arch Dis Child."},{"key":"ref-38","doi-asserted-by":"publisher","first-page":"356-362","DOI":"10.1136\/archdischild-2017-313777","article-title":"Early academic 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status","group":{"name":"current-referee-status","label":"Current Referee Status"}},{"value":"10.3310\/nihropenres.14936.r33453, Melanie Ehrler, University Children's Hospital Zurich, Zurich, 28 Nov 2024, version 1, indexed","URL":"https:\/\/openresearch.nihr.ac.uk\/articles\/4-68\/v1#referee-response-33453","order":0,"name":"referee-response-33453","label":"Referee Report","group":{"name":"article-reports","label":"Article Reports"}},{"value":"10.3310\/nihropenres.14936.r33447, Michael Fleming, University of Glasgow, Glasgow, Scotland, 26 Dec 2024, version 1, indexed","URL":"https:\/\/openresearch.nihr.ac.uk\/articles\/4-68\/v1#referee-response-33447","order":1,"name":"referee-response-33447","label":"Referee Report","group":{"name":"article-reports","label":"Article Reports"}},{"value":"10.3310\/nihropenres.14936.r34057, Kirti Naranje, Sanjay Gandhi Postgraduate Institute of Medical Sciences, Lucknow, 02 Jan 2025, version 1, indexed","URL":"https:\/\/openresearch.nihr.ac.uk\/articles\/4-68\/v1#referee-response-34057","order":2,"name":"referee-response-34057","label":"Referee Report","group":{"name":"article-reports","label":"Article Reports"}},{"value":"This project is funded by the National Institute for Health Research (NIHR) under its \u2018Programme Grants for Applied Research\u2019 (Grant Reference Number NIHR202025). RG is supported by a NIHR Senior Investigator award. ECHILD is supported by ADR UK (Administrative Data Research UK), and the Economic and Social Research Council (part of UK Research and Innovation) (ES\/V000977\/1, ES\/X003663\/1, ES\/X000427\/1). Research at UCL Great Ormond Street Institute of Child Health is supported by the NIHR Great Ormond Street Hospital Biomedical Research Centre. The views expressed are those of the author(s) and not necessarily those of the NIHR or the Department of Health and Social Care. The funders had no role in study design, data collection and analysis, decision to publish, or preparation of the manuscript. The funders had no role in study design, data collection and analysis, decision to publish, or preparation of the manuscript.","order":3,"name":"grant-information","label":"Grant Information"},{"value":"This is an open access article distributed under the terms of the Creative Commons Attribution Licence, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.","order":0,"name":"copyright-info","label":"Copyright"}]}}