Evidence map›Paper›PMID 38496825›Full record

ArticleAnnals of the Child Neurology Society2023

Distribution of hand function by age in individuals with Rett syndrome.

Jeffrey L Neul, Tim A Benke, Eric D Marsh, Jane B Lane, David N Lieberman, Steven A Skinner, Daniel G Glaze, Bernhard Suter, Peter T Heydemann, Arthur A Beisang and 6 more

Abstract read
In one paragraph

Article in Annals of the Child Neurology Society, 2023. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 8 papers.

0numbers the graph read from it
0cells of the map it votes in
8citing papers in PubMed
–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

8 citing papers in PubMed.

  1. Article
  2. Article
  3. Article
  4. Rett syndrome.Nature reviews. Disease primers · 2024
    Review
  5. Review
  6. Rett syndrome: The Natural History Study journey.Annals of the Child Neurology Society · 2024
    Review
  7. Article
  8. Rett syndrome in Ireland: a demographic study.Orphanet journal of rare diseases · 2024
    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.

Jeffrey L NeulVanderbilt University Medical School, Nashville, TN.ORCID 0000-0002-5628-5872
Tim A BenkeUniversity of Colorado, School of Medicine, Children's Hospital Colorado, Aurora, CO.
Eric D MarshChildren's Hospital of Philadelphia, University of Pennsylvania Perelman School of Medicine, Philadelphia, PA.
Jane B LaneUniversity of Alabama at Birmingham, Birmingham, AL.
David N LiebermanBoston Children's Hospital, Harvard University, Boston, MA.
Steven A SkinnerGreenwood Genetic Center, Greenwood, SC.
Daniel G GlazeBaylor College of Medicine, Houston, TX.
Bernhard SuterBaylor College of Medicine, Houston, TX.
Peter T HeydemannRush Medical Center, Chicago, IL.
Arthur A BeisangGillette Specialty Healthcare, St. Paul, MN.
Shannon M StandridgeCincinnati Children's Hospital, Cincinnati, OH.
Robin C C RytherWashington University, School of Medicine, St. Louis, MO.
Richard H HaasUniversity of California San Diego, San Diego, CA.
Lloyd J EdwardsUniversity of Alabama at Birmingham, School of Public Health, Birmingham, AL.
Amitha AnanthUniversity of Alabama at Birmingham, Birmingham, AL.
Alan K PercyUniversity of Alabama at Birmingham, Birmingham, AL.ORCID 0000-0002-9873-5472

Funding

Vanderbilt Institute for Clinical and Translational Research (VICTR) -Identifying correlates of functional immunity in SARS-CoV-2 convalescent plasmaUL1TR002243 · NCATS · VANDERBILT UNIVERSITY MEDICAL CENTER · PI Paul A. Harris, Wesley H Self · 2017 to 2026
$130.7M
The Vanderbilt Institute for Clinical and Translational Research (VICTR)UL1TR000445 · NCATS · VANDERBILT UNIVERSITY MEDICAL CENTER · PI BERNARD, GORDON RAPHAEL · 2012 to 2016
$41.4M
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 NEUL, JEFFREY L · 2015 to 2019
$7.1M
Methods and Health Informatics CoreP30AR072583 · NIAMS · UNIVERSITY OF ALABAMA AT BIRMINGHAM · PI JEFFREY R. CURTIS · 2020 to 2026
$5.7M
Neurobehavioral and biochemical outcome measures in Rett syndrome rodent modelsR01HD083181 · NICHD · BAYLOR COLLEGE OF MEDICINE · PI NEUL, JEFFREY L, SAMACO, RODNEY C · 2016 to 2020
$3.0M
NCATS NIH HHS UL1 TR000445NCATS NIH HHS UL1 TR002243NIAMS NIH HHS P30 AR072583NICHD NIH HHS P50 HD103537NICHD NIH HHS R01 HD083181NICHD NIH HHS U54 HD061222NICHD NIH HHS U54 HD083211
6 · The paper itself

Abstract

Objective: To determine the longitudinal distribution of hand function skills in individuals with classic Rett Syndrome (RTT), an X-linked dominant neurodevelopmental disorder, and correlate with Method: We conducted a longitudinal study of 946 girls and young women with typical RTT seen between 2006 and 2021 in the US Natural History Study (NHS) featuring a structured clinical evaluation to assess the level of hand function skills. The specific focus in this study was to assess longitudinal variation of hand skills from age 2 through age 18 years in relation to specific Results: Following the initial regression period, hand function continues to decline across the age spectrum in individuals with RTT. Specific differences are noted with steeper declines in hand function among those with milder variants (Group A: R133C, R294X, R306C, and C-terminal truncations) compared to groups composed of individuals with more severe variants. Conclusions: These temporal variations in hand use represent specific considerations which could influence the design of clinical trials that test therapies aiming to ameliorate specific functional limitations in individuals with RTT. Furthermore, the distinct impact of specific

Indexed as

hand functionlongitudinal assessmentnatural historyRett syndrome

Identifiers

PMID38496825
PMCPMC10939125

What OpenQuestion holds

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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.