Evidence map›Paper›PMID 32547374›Full record

ArticleFrontiers in integrative neuroscience2020

Evoked Potentials and EEG Analysis in Rett Syndrome and Related Developmental Encephalopathies: Towards a Biomarker for Translational Research.

Joni N Saby, Sarika U Peters, Timothy P L Roberts, Charles A Nelson, Eric D Marsh

Registry-linked trialOpen access · goldAbstract read
In one paragraph

Article in Frontiers in integrative neuroscience, 2020. The graph could read no effect estimate from its abstract, so it casts no vote on the map. It is linked to trial NCT05932589 (Characterization of Translatable Neurophysiological Biomarkers to Enhance Therapeutic Development in Rett Syndrome), which is not on this map. Cited by 27 papers, 2 of them syntheses that pooled it.

0numbers the graph read from it
0cells of the map it votes in
27citing papers in PubMed, 2 pooled it
3.2field-weighted citation impact, top 8% 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.

NCT05932589 recruitingnot on this mapstarted 2023, after this paper: background citation

Characterization of Translatable Neurophysiological Biomarkers to Enhance Therapeutic Development in Rett Syndrome

TypeobservationalSponsorChildren's Hospital of PhiladelphiaRan2023 to 2029Enrolled202ConditionsRett Syndrome, RTT, Rett Syndrome, AtypicalArmsEEG and Auditory and Visual Evoked Potentials (AEP and VEP), Clinical assessment
3 · Its place in the literature

Who cites it

27 citing papers in PubMed, 2 syntheses or guidelines pooled it, 46 citations in OpenAlex.

  1. Auditory processing in rodent models of autism: a systematic review.Journal of neurodevelopmental disorders · 2022
    Pooled it
  2. Pooled it
  3. Review
  4. Visual Recovery Reflects Cortical MeCP2 Sensitivity in Rett Syndrome.Annals of clinical and translational neurology · 2026
    Article
  5. Observational
  6. Article
  7. International journal of molecular sciences · 2025
    Article
  8. Article
  9. Article
  10. Article
  11. Review
  12. Review
  13. Article
  14. Article
  15. Review
  16. Article
  17. Article
  18. Review
  19. Review
  20. Breathing disturbances in Rett syndrome.Handbook of clinical neurology · 2022
    Review
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 4 institutions in 1 country.

Joni N SabyLurie Family Foundations MEG Imaging Center, Department of Radiology, The Children's Hospital of Philadelphia, Philadelphia, PA, United States.
Sarika U PetersDepartment of Pediatrics, Vanderbilt University Medical Center, Nashville, TN, United States.
Timothy P L RobertsLurie Family Foundations MEG Imaging Center, Department of Radiology, The Children's Hospital of Philadelphia, Philadelphia, PA, United States.
Charles A NelsonLaboratories of Cognitive Neuroscience, Division of Developmental Medicine, Boston Children's Hospital, Harvard Medical School, Boston, MA, United States.
Eric D MarshDivision of Neurology and Pediatrics, Children's Hospital of Philadelphia, Philadelphia, PA, United States.
University of Pennsylvania · USHarvard University · USImaging Center · USVanderbilt University Medical Center · US

Funding

Training and Career Development ComponentU54HD061222 · NICHD · UNIVERSITY OF ALABAMA AT BIRMINGHAM · PI PERCY, ALAN KENNETH · 2009 to 2019
$12.4M
Overall: Eunice Kennedy Shriver Intellectual and Developmental Disabilities Research Center at VanderbiltP50HD103537 · NICHD · VANDERBILT UNIVERSITY MEDICAL CENTER · PI Lea K Davis · 2020 to 2026
$10.3M
Research Project: Sensory and Multisensory Contributions to AutismU54HD083211 · NICHD · VANDERBILT UNIVERSITY MEDICAL CENTER · PI DYKENS, ELISABETH MAY · 2015 to 2019
$7.1M
The Intellectual and Developmental Disabilities Research Center at CHOP/PennU54HD086984 · NICHD · CHILDREN'S HOSP OF PHILADELPHIA · PI SCHULTZ, ROBERT THOMAS · 2016 to 2020
$6.3M
Mouse Neurodevelopmental Behavior CoreU54HD090255 · NICHD · BOSTON CHILDREN'S HOSPITAL · PI POMEROY, SCOTT LOREN · 2016 to 2020
$5.2M
Neural Signatures of Tactile Processing and Sensorimotor GABA in Development and Autism Spectrum DisorderK01MH118378 · NIMH · CHILDREN'S HOSP OF PHILADELPHIA · PI SABY, JONI N. · 2019 to 2022
$496k
NICHD NIH HHS P50 HD103537NICHD NIH HHS U54 HD061222NICHD NIH HHS U54 HD083211NICHD NIH HHS U54 HD086984NICHD NIH HHS U54 HD090255NIMH NIH HHS K01 MH118378
6 · The paper itself

Abstract

Rett syndrome is a debilitating neurodevelopmental disorder for which no disease-modifying treatment is available. Fortunately, advances in our understanding of the genetics and pathophysiology of Rett syndrome has led to the development of promising new therapeutics for the condition. Several of these therapeutics are currently being tested in clinical trials with others likely to progress to clinical trials in the coming years. The failure of recent clinical trials for Rett syndrome and other neurodevelopmental disorders has highlighted the need for electrophysiological or other objective biological markers of treatment response to support the success of clinical trials moving forward. The purpose of this review is to describe the existing studies of electroencephalography (EEG) and evoked potentials (EPs) in Rett syndrome and discuss the open questions that must be addressed before the field can adopt these measures as surrogate endpoints in clinical trials. In addition to summarizing the human work on Rett syndrome, we also describe relevant studies with animal models and the limited research that has been carried out on Rett-related disorders, particularly methyl-CpG binding protein 2 (MECP2) duplication syndrome, CDKL5 deficiency disorder, and FOXG1 disorder.

Indexed as

biomarkerdevelopmental encephalopathyEEGevoked potentialRett syndrome

Identifiers

PMID32547374
PMCPMC7271894
OpenAlexW3031077389

What OpenQuestion holds

Textmetadata
LicenceCC BY
Read underepoch 390

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.