Evidence map›Paper›PMID 35675091›Full record

ArticlePaediatric and perinatal epidemiology2022

Temporal and geographical variations in survival of children born with congenital anomalies in Europe: A multi-registry cohort study.

Michele Santoro, Alessio Coi, Anna Pierini, Judith Rankin, Svetlana V Glinianaia, Joachim Tan, Abigail Reid, Ester Garne, Maria Loane, Joanne Given and 26 more

Open access · hybridAbstract read
In one paragraph

Article in Paediatric and perinatal epidemiology, 2022. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 12 papers, 1 of them a synthesis that pooled it.

0numbers the graph read from it
0cells of the map it votes in
12citing papers in PubMed, 1 pooled it
4.9field-weighted citation impact, top 5% of its field
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

12 citing papers in PubMed, 1 synthesis or guideline pooled it, 15 citations in OpenAlex.

  1. Pooled it
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4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

36 authors at 19 institutions in 14 countries.

Michele SantoroUnit of Epidemiology of Rare diseases and Congenital anomalies, Institute of Clinical Physiology, National Research Council, Pisa, Italy.ORCID 0000-0003-0676-3036
Alessio CoiUnit of Epidemiology of Rare diseases and Congenital anomalies, Institute of Clinical Physiology, National Research Council, Pisa, Italy.
Anna PieriniUnit of Epidemiology of Rare diseases and Congenital anomalies, Institute of Clinical Physiology, National Research Council, Pisa, Italy.
Judith RankinPopulation Health Sciences Institute, Faculty of Medical Sciences, Newcastle University, Newcastle upon Tyne, UK.
Svetlana V GlinianaiaPopulation Health Sciences Institute, Faculty of Medical Sciences, Newcastle University, Newcastle upon Tyne, UK.
Joachim TanPopulation Health Research Institute, St George's, University of London, London, UK.
Abigail ReidPopulation Health Research Institute, St George's, University of London, London, UK.
Ester GarnePaediatric Department, Hospital Lillebaelt, Kolding, Denmark.
Maria LoaneFaculty of Life and Health Sciences, Ulster University, Coleraine, UK.
Joanne GivenFaculty of Life and Health Sciences, Ulster University, Coleraine, UK.ORCID 0000-0003-4921-1944
Amaia AizpuruaPublic Health Division of Gipuzkoa, BioDonostia Research Institute, San Sebastian, Spain.
Gianni AstolfiIMER Registry, Department of Neuroscience and Rehabilitation, University of Ferrara, Ferrara, Italy.
Ingeborg BarisicChildren's Hospital Zagreb, Centre of Excellence for Reproductive and Regenerative Medicine, Medical School University of Zagreb, Zagreb, Croatia.
Clara Cavero-CarbonellRare Diseases Research Unit, Foundation for the Promotion of Health and Biomedical Research in the Valencian Region, Valencia, Spain.
Hermien E K de WalleDepartment of Genetics, University Medical Center Groningen, University of Groningen, Groningen, the Netherlands.
Elly Den HondProvincial Institute for Hygiene, Antwerp, Belgium.
Laura García-VillodreRare Diseases Research Unit, Foundation for the Promotion of Health and Biomedical Research in the Valencian Region, Valencia, Spain.
Miriam GattMalta Congenital Anomalies Registry, Directorate for Health Information and Research, Pieta, Malta.
Mika GisslerTHL Finnish Institute for Health and Welfare, Information Services Department, Helsinki, Finland.
Sue JordanFaculty of Medicine, Health & Life Science, Swansea University, Swansea, UK.
Babak KhoshnoodUniversité de Paris, CRESS-Epopé, INSERM, INRA, Paris, France.ORCID 0000-0002-4031-4915
Sonja Kiuru-KuhlefeltTHL Finnish Institute for Health and Welfare, Information Services Department, Helsinki, Finland.
Kari KlungsøyrDepartment of Global Public Health and Primary Care, University of Bergen, Bergen, Norway.
Nathalie LelongUniversité de Paris, CRESS-Epopé, INSERM, INRA, Paris, France.
Renée LutkeDepartment of Genetics, University Medical Center Groningen, University of Groningen, Groningen, the Netherlands.
Olatz MokoroaPublic Health Division of Gipuzkoa, BioDonostia Research Institute, San Sebastian, Spain.ORCID 0000-0003-3831-6089
Vera NelenProvincial Institute for Hygiene, Antwerp, Belgium.
Amanda J NevilleImer registry Centre for Epidemiology and Clinical Research University of Ferrara and Azienda Ospedaliera Universitaria di Ferrara, Ferrara, Italy.
Ljubica OdakChildren's Hospital Zagreb, Centre of Excellence for Reproductive and Regenerative Medicine, Medical School University of Zagreb, Zagreb, Croatia.
Anke RissmannMalformation Monitoring Centre Saxony-Anhalt, Medical Faculty Otto-von-Guericke-University Magdeburg, Magdeburg, Germany.ORCID 0000-0002-9437-2790
Ieuan ScanlonFaculty of Medicine, Health & Life Science, Swansea University, Swansea, UK.
Stine Kjaer UrhojPaediatric Department, Hospital Lillebaelt, Kolding, Denmark.
Diana WellesleyFaculty of Medicine, University of Southampton and Wessex Clinical Genetics Service, Princess Anne Hospital, Southampton, UK.
Wladimir WerteleckiOMNI-Net for Children International Charitable Fund, Rivne, Ukraine.
Lyubov YevtushokOMNI-Net for Children International Charitable Fund, Rivne, Ukraine.
Joan K MorrisPopulation Health Research Institute, St George's, University of London, London, UK.ORCID 0000-0002-7164-612X
Istituto di Fisiologia Clinica · ITSt George's, University of London · GBBiogipuzkoa Health Research Institute · ESFinnish Institute for Health and Welfare · FIFundación para el Fomento de la Investigación Sanitaria y Biomédica de la Comunitat Valenciana · ESInserm · FRLillebaelt Hospital · DKNewcastle University · GBProvincial Institute for Hygiene · BERivne State University of Humanities · UASwansea University · GBUniversity Hospital Centre Zagreb · HRUniversity Medical Center Groningen · NLUniversity of Ferrara · ITUniversity of Ulster · GBMinistry for Health · MTNorwegian Institute of Public Health · NOOtto-von-Guericke-Universität Magdeburg · DEPrincess Anne Hospital · GB

Funding

Medical Research Council MR/K02325X/1
6 · The paper itself

Abstract

backgroundCongenital anomalies are a major cause of perinatal, neonatal and infant mortality.

objectivesThe aim was to investigate temporal changes and geographical variation in survival of children with major congenital anomalies (CA) in different European areas.

methodsIn this population-based linkage cohort study, 17 CA registries members of EUROCAT, the European network for the surveillance of CAs, successfully linked data on 115,219 live births with CAs to mortality records. Registries estimated Kaplan-Meier survival at 28 days and 5 years of age and fitted Cox's proportional hazards models comparing mortality at 1 year and 1-9 years of age for children born during 2005-2014 with those born during 1995-2004. The hazard ratios (HR) from each registry were combined centrally using a random-effects model. The 5-year survival conditional on having survived to 28 days of age was calculated.

resultsThe overall risk of death by 1 year of age for children born with any major CA in 2005-2014 decreased compared to 1995-2004 (HR 0.68, 95% confidence interval [CI] 0.53, 0.89). Survival at 5 years of age ranged between registries from 97.6% to 87.0%. The lowest survival was observed for the registry of OMNI-Net (Ukraine) (87.0%, 95% CI 86.1, 87.9).

conclusionsSurvival of children with CAs improved for births in 2005-2014 compared with 1995-2004. The use of CA registry data linked to mortality data enables investigation of survival of children with CAs. Factors such as defining major CAs, proportion of terminations of pregnancy for foetal anomaly, source of mortality data and linkage methods are important to consider in the design of future studies and in the interpretation of the results on survival of children with CAs.

Indexed as

Congenital AbnormalitiesParturitionChildCohort StudiesEuropeFemaleHumansInfantInfant MortalityInfant, NewbornPregnancyPrevalenceRegistriescohort studycongenital anomaliesregistrysurvival

Identifiers

PMID35675091
PMCPMC9796712
OpenAlexW4282570422

What OpenQuestion holds

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LicenceCC BY-NC-ND
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Registered trials

None linked

Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.