Evidence map›Paper›PMID 34687210›Full record

ArticleBrain : a journal of neurology2021

Early childhood epilepsies: epidemiology, classification, aetiology, and socio-economic determinants.

Joseph D Symonds, Katherine S Elliott, Jay Shetty, Martin Armstrong, Andreas Brunklaus, Ioana Cutcutache, Louise A Diver, Liam Dorris, Sarah Gardiner, Alice Jollands and 13 more

Registry-linked trialOpen access · bronzeAbstract read
In one paragraph

Article in Brain : a journal of neurology, 2021. The graph could read no effect estimate from its abstract, so it casts no vote on the map. It is linked to trial NCT06223334 (Clinico-etiological Profile of Epileptic Syndromes in Infants and Early Childhood), which is not on this map. Cited by 109 papers, 3 of them syntheses that pooled it.

0numbers the graph read from it
0cells of the map it votes in
109citing papers in PubMed, 3 pooled it
20.0field-weighted citation impact, top 1% 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.

NCT06223334 unknown statusnot on this mapstarted 2023, after this paper: background citation

Clinico-etiological Profile of Epileptic Syndromes in Infants and Early Childhood

TypeobservationalSponsorAl-Azhar UniversityRan2023 to 2025Enrolled140ConditionsEpileptic SyndromesArmsNo intervention
3 · Its place in the literature

Who cites it

109 citing papers in PubMed, 3 syntheses or guidelines pooled it, 219 citations in OpenAlex.

  1. Efficacy and safety of Brivaracetam as adjunctive therapy in pediatric epilepsy: A systematic review and meta-analysis.Neurological sciences : official journal of the Italian Neurological Society and of the Italian Society of Clinical Neurophysiology · 2025
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  11. The Bright Side of the Spectrum: Self-Limited Infantile Epilepsy-Related SCN8A Gof Variant.International journal of developmental neuroscience : the official journal of the International Society for Developmental Neuroscience · 2026
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  12. Bayesian age-period-cohort analysis and trend prediction of epilepsy disease burden in China, 1990-2021.Epileptic disorders : international epilepsy journal with videotape · 2026
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49 more citing papers are in PubMed but not listed here.

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

23 authors at 9 institutions in 3 countries.

Joseph D SymondsPaediatric Neurosciences Research Group, Royal Hospital for Children, Glasgow G51 4TF, UK.ORCID 0000-0002-2141-4216
Katherine S ElliottWellcome Centre for Human Genetics, University of Oxford, Oxford OX3 7BN, UK.
Jay ShettyDepartment of Paediatric Neurosciences, Royal Hospital for Sick Children, Sciennes Road, Edinburgh EH9 1LF, UK.ORCID 0000-0002-3114-4002
Martin ArmstrongUCB Pharma, Braine l'Alleud, Belgium.
Andreas BrunklausPaediatric Neurosciences Research Group, Royal Hospital for Children, Glasgow G51 4TF, UK.
Ioana CutcutacheUCB Pharma, Slough, UK.
Louise A DiverWest of Scotland Regional Genetics Service, Queen Elizabeth University Hospitals, Glasgow G51 4TF, UK.
Liam DorrisPaediatric Neurosciences Research Group, Royal Hospital for Children, Glasgow G51 4TF, UK.
Sarah GardinerWest of Scotland Regional Genetics Service, Queen Elizabeth University Hospitals, Glasgow G51 4TF, UK.
Alice JollandsPaediatric Neurology, Tayside Children's Hospital, Dundee DD1 9SY, UK.
Shelagh JossWest of Scotland Regional Genetics Service, Queen Elizabeth University Hospitals, Glasgow G51 4TF, UK.
Martin KirkpatrickPaediatric Neurology, Tayside Children's Hospital, Dundee DD1 9SY, UK.
Ailsa McLellanDepartment of Paediatric Neurosciences, Royal Hospital for Sick Children, Sciennes Road, Edinburgh EH9 1LF, UK.
Stewart MacLeodPaediatric Neurosciences Research Group, Royal Hospital for Children, Glasgow G51 4TF, UK.
Mary O'ReganPaediatric Neurosciences Research Group, Royal Hospital for Children, Glasgow G51 4TF, UK.
Matthew PageUCB Pharma, Braine l'Alleud, Belgium.
Elizabeth PilleyDepartment of Paediatric Neurosciences, Royal Hospital for Sick Children, Sciennes Road, Edinburgh EH9 1LF, UK.
Daniela T PilzWest of Scotland Regional Genetics Service, Queen Elizabeth University Hospitals, Glasgow G51 4TF, UK.
Elma StephenPaediatric Neurology, Royal Aberdeen Children's Hospital, Aberdeen AB25 2ZG, UK.
Kirsty StewartWest of Scotland Regional Genetics Service, Queen Elizabeth University Hospitals, Glasgow G51 4TF, UK.
Houman AshrafianDivision of Cardiovascular Medicine, John Radcliffe Hospital, Oxford, UK.
Julian C KnightWellcome Centre for Human Genetics, University of Oxford, Oxford OX3 7BN, UK.
Sameer M ZuberiPaediatric Neurosciences Research Group, Royal Hospital for Children, Glasgow G51 4TF, UK.
Royal Hospital for Children · GBQueen Elizabeth University Hospital · GBCentre for Human Genetics · GBUCB Pharma (Belgium) · BEJohn Radcliffe Hospital · GBNHS Tayside · GBRoyal Aberdeen Children's Hospital · GBUCB Pharma (United Kingdom) · GBUniversity of Dundee · GB

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Epilepsies of early childhood are frequently resistant to therapy and often associated with cognitive and behavioural comorbidity. Aetiology focused precision medicine, notably gene-based therapies, may prevent seizures and comorbidities. Epidemiological data utilizing modern diagnostic techniques including whole genome sequencing and neuroimaging can inform diagnostic strategies and therapeutic trials. We present a 3-year, multicentre prospective cohort study, involving all children under 3 years of age in Scotland presenting with epilepsies. We used two independent sources for case identification: clinical reporting and EEG record review. Capture-recapture methodology was then used to improve the accuracy of incidence estimates. Socio-demographic and clinical details were obtained at presentation, and 24 months later. Children were extensively investigated for aetiology. Whole genome sequencing was offered for all patients with drug-resistant epilepsy for whom no aetiology could yet be identified. Multivariate logistic regression modelling was used to determine associations between clinical features, aetiology, and outcome. Three hundred and ninety children were recruited over 3 years. The adjusted incidence of epilepsies presenting in the first 3 years of life was 239 per 100 000 live births [95% confidence interval (CI) 216-263]. There was a socio-economic gradient to incidence, with a significantly higher incidence in the most deprived quintile (301 per 100 000 live births, 95% CI 251-357) compared with the least deprived quintile (182 per 100 000 live births, 95% CI 139-233), χ2 odds ratio = 1.7 (95% CI 1.3-2.2). The relationship between deprivation and incidence was only observed in the group without identified aetiology, suggesting that populations living in higher deprivation areas have greater multifactorial risk for epilepsy. Aetiology was determined in 54% of children, and epilepsy syndrome was classified in 54%. Thirty-one per cent had an identified genetic cause for their epilepsy. We present novel data on the aetiological spectrum of the most commonly presenting epilepsies of early childhood. Twenty-four months after presentation, 36% of children had drug-resistant epilepsy (DRE), and 49% had global developmental delay (GDD). Identification of an aetiology was the strongest determinant of both DRE and GDD. Aetiology was determined in 82% of those with DRE, and 75% of those with GDD. In young children with epilepsy, genetic testing should be prioritized as it has the highest yield of any investigation and is most likely to inform precision therapy and prognosis. Epilepsies in early childhood are 30% more common than previously reported. Epilepsies of undetermined aetiology present more frequently in deprived communities. This likely reflects increased multifactorial risk within these populations.

Indexed as

Socioeconomic FactorsCausalityChild, PreschoolCohort StudiesDrug Resistant EpilepsyEpilepsyFemaleFollow-Up StudiesHumansInfantInfant, NewbornMaleProspective StudiesRetrospective StudiesScotlandepidemiologyepilepsygeneticsincidenceprecision

Identifiers

PMID34687210
PMCPMC8557326
OpenAlexW3168961767

What OpenQuestion holds

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LicenceCC BY-NC
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Registered trials

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.