Evidence map›Paper›PMID 28457992›Full record

Trial reportEpilepsy & behavior : E&B2017

Influence of seizures on early development in tuberous sclerosis complex.

Jamie K Capal, Beatriz Bernardino-Cuesta, Paul S Horn, Donna Murray, Anna Weber Byars, Nicole M Bing, Bridget Kent, Deborah A Pearson, Mustafa Sahin, Darcy A Krueger and 1 more

Abstract readClinical TrialMulticenter Study
In one paragraph

Trial report in Epilepsy & behavior : E&B, 2017. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 91 papers, 4 of them syntheses that pooled it.

0numbers the graph read from it
0cells of the map it votes in
91citing papers in PubMed, 4 pooled it
–field-weighted citation impact
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

91 citing papers in PubMed, 4 syntheses or guidelines pooled it.

  1. Pooled it
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  5. Trial
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  8. Is autism driven by epilepsy in infants with Tuberous Sclerosis Complex?Annals of clinical and translational neurology · 2020
    Trial
  9. Trial
  10. Neonatal Seizures in Tuberous Sclerosis Complex: A Case Series.Annals of the Child Neurology Society · 2026
    Article
  11. Tuberous sclerosis complex.Nature reviews. Disease primers · 2026
    Review
  12. Article
  13. Article
  14. Article
  15. Article
  16. Review
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31 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

11 authors.

Jamie K CapalDepartment of Neurology MLC 2015, Cincinnati Children's Hospital Medical Center, 3333 Burnet Avenue, Cincinnati, OH 45229, USA. Electronic address: Jamie.capal@cchmc.org.
Beatriz Bernardino-CuestaSeccion de Neuropediatria, Hospital Infantil Universitario Nino Jesus, Madrid, Spain.
Paul S HornDepartment of Neurology MLC 2015, Cincinnati Children's Hospital Medical Center, 3333 Burnet Avenue, Cincinnati, OH 45229, USA.
Donna MurrayDepartment of Developmental and Behavioral Pediatrics, Cincinnati Children's Hospital Medical Center, 3430 Burnet Avenue, Cincinnati, OH 45229, USA; Autism Speaks Inc, 85 Devonshire St, Boston, MA 02109, USA.
Anna Weber ByarsDepartment of Neurology MLC 2015, Cincinnati Children's Hospital Medical Center, 3333 Burnet Avenue, Cincinnati, OH 45229, USA.
Nicole M BingDepartment of Developmental and Behavioral Pediatrics, Cincinnati Children's Hospital Medical Center, 3430 Burnet Avenue, Cincinnati, OH 45229, USA.
Bridget KentDepartment of Developmental and Behavioral Pediatrics, Cincinnati Children's Hospital Medical Center, 3430 Burnet Avenue, Cincinnati, OH 45229, USA.
Deborah A PearsonMcGovern Medical School, The University of Texas Health Science Center at Houston, 6431 Fannin Street, Houston, TX 77030, USA.
Mustafa SahinDepartment of Neurology, Boston Children's Hospital, 300 Longwood Avenue, Boston, MA 02115, USA.
Darcy A KruegerDepartment of Neurology MLC 2015, Cincinnati Children's Hospital Medical Center, 3333 Burnet Avenue, Cincinnati, OH 45229, USA.
TACERN Study Group

Funding

UCLA Clinical and Translational Science InstituteUL1TR000124 · NCATS · UNIVERSITY OF CALIFORNIA LOS ANGELES · PI DUBINETT, STEVEN M. · 2012 to 2015
$57.0M
Toward better characterization and clinical trial readiness for targeting neuropsychiatric manifestations in PTEN pathogenic variantsU54NS092090 · NINDS · BOSTON CHILDREN'S HOSPITAL · PI M. Pilar Trelles · 2014 to 2026
$20.1M
Early Biomarkers of Autism Spectrum Disorders in infants with Tuberous SclerosisU01NS082320 · NINDS · BOSTON CHILDREN'S HOSPITAL · PI KRUEGER, DARCY, SAHIN, MUSTAFA · 2012 to 2018
$13.3M
Cincinnati Center for Clinical and Translational Sciences and TrainingUL1TR000077 · NCATS · UNIVERSITY OF CINCINNATI · PI HEUBI, JAMES E. · 2012 to 2014
$9.9M
Preventing Epilepsy using Vigabatrin in Infants with Tuberous Sclerosis ComplexU01NS092595 · NINDS · UNIVERSITY OF ALABAMA AT BIRMINGHAM · PI BEBIN, MARTINA · 2016 to 2020
$7.1M
Mouse Neurodevelopmental Behavior CoreU54HD090255 · NICHD · BOSTON CHILDREN'S HOSPITAL · PI POMEROY, SCOTT LOREN · 2016 to 2020
$5.2M
High Frequency Oscillation as a Biomarker in Childhood EpilepsyR01NS082649 · NINDS · UNIVERSITY OF CALIFORNIA LOS ANGELES · PI WU, JOYCE Y · 2013 to 2017
$1.7M
Potential EEG biomarkers and antiepileptogenic strategies for epilepy in TSCP20NS080199 · NINDS · UNIVERSITY OF ALABAMA AT BIRMINGHAM · PI BEBIN, MARTINA · 2012 to 2014
$966k
NCATS NIH HHS UL1 TR000077NCATS NIH HHS UL1 TR000124NICHD NIH HHS U54 HD090255NINDS NIH HHS P20 NS080199NINDS NIH HHS R01 NS082649NINDS NIH HHS U01 NS082320NINDS NIH HHS U01 NS092595NINDS NIH HHS U54 NS092090
6 · The paper itself

Abstract

objectiveEpilepsy is commonly seen in Tuberous Sclerosis Complex (TSC). The relationship between seizures and developmental outcomes has been reported, but few studies have examined this relationship in a prospective, longitudinal manner. The objective of the study was to evaluate the relationship between seizures and early development in TSC.

methodsAnalysis of 130 patients ages 0-36months with TSC participating in the TSC Autism Center of Excellence Network, a large multicenter, prospective observational study evaluating biomarkers predictive of autism spectrum disorder (ASD), was performed. Infants were evaluated longitudinally with standardized evaluations, including cognitive, adaptive, and autism-specific measures. Seizure history was collected continuously throughout, including seizure type and frequency.

resultsData were analyzed at 6, 12, 18, and 24months of age. Patients without a history of seizures performed better on all developmental assessments at all time points compared to patients with a history of seizures and exhibited normal development at 24months. Patients with a history of seizures not only performed worse, but developmental progress lagged behind the group without seizures. All patients with a history of infantile spasms performed worse on all developmental assessments at 12, 18, and 24months. Higher seizure frequency correlated with poorer outcomes on developmental testing at all time points, but particularly at 12months and beyond. Patients with higher seizure frequency during infancy continued to perform worse developmentally through 24months. A logistic model looking at the individual impact of infantile spasms, seizure frequency, and age of seizure onset as predictors of developmental delay revealed that age of seizure onset was the most important factor in determining developmental outcome.

conclusionsResults of this study further define the relationship between seizures and developmental outcomes in young children with TSC. Early seizure onset in infants with TSC negatively impacts very early neurodevelopment, which persists through 24months of age.

Indexed as

Child, PreschoolDevelopmental DisabilitiesFemaleHumansInfantLongitudinal StudiesMaleProspective StudiesSeizuresSpasms, InfantileTuberous SclerosisAutism spectrum disorderCognitive developmentEpilepsyInfantile spasmsSeizuresTuberous Sclerosis Complex

Identifiers

PMID28457992
PMCPMC5497719

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Registered trials

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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.