Evidence map›Paper›PMID 36372969›Full record

ArticleAmerican journal of medical genetics. Part A2023

Analysis of electrocardiograms in individuals with CDKL5 deficiency disorder.

Jessica Stansauk, Andrea Fidell, Tim Benke, Michael Schaffer, Scott T Demarest

Open access · greenAbstract read
In one paragraph

Article in American journal of medical genetics. Part A, 2023. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 3 papers.

0numbers the graph read from it
0cells of the map it votes in
3citing papers in PubMed
0.9field-weighted citation impact, top 22% 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

3 citing papers in PubMed, 5 citations in OpenAlex.

  1. Transcriptomic Profiling of Zebrafish Mutant forInternational journal of molecular sciences · 2025
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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

5 authors at 1 institution in 1 country.

Jessica StansaukSection of Genetics and Metabolism, University of Colorado School of Medicine, Children's Hospital Colorado, Aurora, Colorado, USA.ORCID https://orcid.org/0000-0003-4727-9718
Andrea FidellSection of Neurology, University of Colorado School of Medicine, Children's Hospital Colorado, Aurora, Colorado, USA.
Tim BenkeSection of Neurology, University of Colorado School of Medicine, Children's Hospital Colorado, Aurora, Colorado, USA.
Michael SchafferSection of Cardiology/Department of Pediatrics, University of Colorado School of Medicine, Children's Hospital Colorado, Aurora, Colorado, USA.
Scott T DemarestSection of Neurology, University of Colorado School of Medicine, Children's Hospital Colorado, Aurora, Colorado, USA.
Children's Hospital Colorado · US

Funding

Training and Career Development ComponentU54HD061222 · NICHD · UNIVERSITY OF ALABAMA AT BIRMINGHAM · PI PERCY, ALAN KENNETH · 2009 to 2019
$12.4M
Multi-Site Validation of Biomarkers and Core Clinical Outcome Measures for Clinical Trials Readiness in CDKL5 Deficiency DisorderU01NS114312 · NINDS · UNIVERSITY OF COLORADO DENVER · PI BENKE, TIMOTHY A, DOWNS, JENNY · 2021 to 2025
$4.7M
International Foundation for CDKL5 Research, NIH-NICHD Rett-Related Disorders Natural History Study consortiumNICHD NIH HHS U54 HD061222NIH-NINDS Multisite validation of biomarkers and core clinical outcome measures for clinical trials readiness in CDKL5 Deficiency Disorder U01NS114312NINDS NIH HHS U01 NS114312
6 · The paper itself

Abstract

CDKL5 deficiency disorder (CDD) is an epileptic encephalopathy associated with medically refractory epilepsy. We sought to determine whether prolonged corrected QT interval (QTc) or other cardiac conduction abnormalities were seen in CDD in a clinical cohort. A cohort of individuals with CDD was evaluated in the Children's Hospital Colorado's International Foundation for CDKL5 Research designated Center of Excellence clinic with routine electrocardiograms obtained as part of routine clinical care. Retrospective review of electrocardiograms was completed. ECGs from 44 individuals (7 male, 37 female, age range 0-34.5 years) with pathogenic mutations and findings consistent with CDD were evaluated. Multiple ECGs were available from the 44 individuals obtained from 1996 to 2020. Prolonged QTc was found in two individuals (4.5%) and either resolved or was not confirmed on Holter monitor; no additional interventions were performed. A total of 11 individuals had echocardiograms for a variety of indications including unexplained tachycardia and ECG abnormalities; all were normal. Two individuals in the cohort died during the study with no abnormal findings on ECG. The incidence of prolonged QTc or other significant actionable cardiac abnormalities was rare in a cohort of individuals with CDD though was higher than the prevalence seen within the general population. Further studies in a larger, confirmatory cohort over a longer period are needed.

Indexed as

Epileptic SyndromesLong QT SyndromeSpasms, InfantileAdolescentAdultChildChild, PreschoolElectrocardiographyFemaleHumansInfantInfant, NewbornMaleProtein Serine-Threonine KinasesYoung AdultCDKL5 protein, humanProtein Serine-Threonine KinasesCDDCDKL5long QT

Identifiers

PMID36372969
PMCPMC9886132
OpenAlexW4308957365

What OpenQuestion holds

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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.