Evidence map›Paper›PMID 37160334›Full record

SynthesisArchives of disease in childhood2023

Survival, hospitalisation and surgery in children born with Pierre Robin sequence: a European population-based cohort study.

Michele Santoro, Ester Garne, Alessio Coi, Joachim Tan, Maria Loane, Elisa Ballardini, Clara Cavero-Carbonell, Hermien Ek de Walle, Miriam Gatt, Mika Gissler and 6 more

Open access · hybridAbstract readMeta-Analysis
In one paragraph

Synthesis in Archives of disease in childhood, 2023. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 1 paper.

0numbers the graph read from it
0cells of the map it votes in
1citing papers in PubMed
0.6field-weighted citation impact, top 24% 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

1 citing paper in PubMed, 2 citations in OpenAlex.

  1. Article
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

16 authors at 13 institutions in 10 countries.

Michele SantoroUnit of Epidemiology of rare Diseases and Congenital Anomalies, Institute of Clinical Physiology, National Research Council, Pisa, Italy msantoro@ifc.cnr.it.ORCID 0000-0003-0676-3036
Ester GarneDepartment of Paediatrics and Adolescent Medicine, Lillebaelt Hospital, University Hospital of Southern Denmark, Kolding, Denmark.
Alessio CoiUnit of Epidemiology of rare Diseases and Congenital Anomalies, Institute of Clinical Physiology, National Research Council, Pisa, Italy.
Joachim TanPopulation Health Research Institute, St George's, University of London, London, UK.
Maria LoaneFaculty of Life and Health Sciences, Ulster University, Newtowanbbey, UK.
Elisa BallardiniNeonatal Intensive Care Unit, Paediatric Section, IMER Registry (Emilia Romagna Registry of Birth Defects), Department of Medical Sciences, University of Ferrara, Ferrara, Italy.
Clara Cavero-CarbonellRare Diseases Research Unit, Foundation for the Promotion of Health and Biomedical Research in the Valencian Region FISABIO, Valencia, Spain.
Hermien Ek de WalleGenetics, University Medical Center Groningen, University of Groningen, Groningen, The Netherlands.
Miriam GattDirectorate for Health Information and Research, Malta Congenital Anomalies Registry, G'mangia, Malta.
Mika GisslerDepartment of Knowledge Brokers, THL Finnish Institute for Health and Welfare, Helsinki, Finland.
Sue JordanFaculty of Medicine, Health and Life Science, Swansea University, Swansea, UK.
Kari KlungsøyrDepartment of Global Public Health and Primary Care, University of Bergen, Bergen, Norway.
Natalie LelongUniversité de Paris, CRESS-Epopé, INSERM, INRA, Paris, France.
Stine Kjaer UrhojDepartment of Paediatrics and Adolescent Medicine, Lillebaelt Hospital, University Hospital of Southern Denmark, Kolding, Denmark.ORCID 0000-0002-2069-9723
Diana G WellesleyFaculty of Medicine, University of Southampton and Wessex Clinical Genetics Service, Princess Anne Hospital, Southampton, UK.
Joan K MorrisPopulation Health Research Institute, St George's, University of London, London, UK.ORCID 0000-0002-7164-612X
Istituto di Fisiologia Clinica · ITLillebaelt Hospital · DKSt George's, University of London · GBFinnish Institute for Health and Welfare · FIFundación para el Fomento de la Investigación Sanitaria y Biomédica de la Comunitat Valenciana · ESInserm · FRMinistry for Health · MTNorwegian Institute of Public Health · NOPrincess Anne Hospital · GBSwansea University · GBUniversity Medical Center Groningen · NLUniversity of Ferrara · ITUniversity of Ulster · GB

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

objectiveTo evaluate survival, hospitalisations and surgical procedures for children born with Pierre Robin sequence (PRS) across Europe.

designMulticentre population-based cohort study.

settingData on 463 live births with PRS from a population of 4 984 793 from 12 EUROCAT congenital anomaly registries.

methodsData on children with PRS born 1995-2014 were linked electronically to data on mortality, hospitalisations and surgical procedures up to 10 years of age. Each registry applied a common data model to standardise the linked data and ran common syntax scripts to produce aggregate tables. Results from each registry were pooled using random-effect meta-analyses.

main outcome measuresProbability of survival, proportion of children hospitalised and undergoing surgery, and median length of hospital stay.

resultsThe majority of deaths occurred in the first year of life with a survival rate of 96.0% (95% CI 93.5% to 98.5%); 95.1% (95% CI 92.7% to 97.7%) survived to age 10. In the first year of life, 99.2% (95% CI 95.0% to 99.9%) of children were hospitalised with a median stay of 21.4 days (95% CI 15.6 to 27.2), and 67.6% (95% CI 46.6% to 81.8%) underwent surgery. In the first 5 years of life, 99.2% of children underwent a median of two surgical procedures. Between ages 5 and 9, 58.3% (95% CI 44.7% to 69.7%) were hospitalised with a median annual stay of 0.3 days.

conclusionsChildren with PRS had high mortality and morbidity with long hospital stays in the first year of life, and almost all had surgery before 5 years of age. Survival improved after infancy with fewer hospitalisations after age 5. This study provides reliable estimates of the survival and morbidity of children with PRS for families and healthcare providers.

Indexed as

Pierre Robin SyndromeChildChild, PreschoolCohort StudiesEuropeHumansInfantLength of StayParturitionChild HealthEpidemiologyMortality

Identifiers

PMID37160334
PMCPMC10314079
OpenAlexW4376117834

What OpenQuestion holds

Textmetadata
LicenceCC BY-NC
Read underepoch 390

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.