Evidence map›Paper›PMID 31586494›Full record

ArticleJournal of neurodevelopmental disorders2019

Developmental divergence: motor trajectories in children with fragile X syndrome with and without co-occurring autism.

Elizabeth A Will, Somer L Bishop, Jane E Roberts

Open access · goldAbstract read
In one paragraph

Article in Journal of neurodevelopmental disorders, 2019. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 14 papers, 1 of them a synthesis that pooled it.

0numbers the graph read from it
0cells of the map it votes in
14citing papers in PubMed, 1 pooled it
1.2field-weighted citation impact, top 21% 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

14 citing papers in PubMed, 1 synthesis or guideline pooled it, 26 citations in OpenAlex.

  1. Pooled it
  2. Article
  3. Up-regulation of Minibrain/DYRK1A contributes to macrocephaly and brain overgrowth in aProceedings of the National Academy of Sciences of the United States of America · 2026
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  4. Prelinguistic Communication Complexity of Children With Neurogenetic Syndromes.Journal of speech, language, and hearing research : JSLHR · 2025
    Article
  5. Article
  6. Article
  7. Social Communication Delay in an Unbiased Sample of Preschoolers With theJournal of speech, language, and hearing research : JSLHR · 2024
    Article
  8. Article
  9. Article
  10. Article
  11. Motor Influences on Communication: Comparisons Between Down Syndrome and Fragile X Syndrome.American journal on intellectual and developmental disabilities · 2021
    Article
  12. Article
  13. Article
  14. 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

3 authors at 2 institutions in 1 country.

Elizabeth A WillDepartment of Psychology, University of South Carolina, 1512 Pendleton Street, Columbia, SC, 29208, USA. willea@mailbox.sc.edu.ORCID 0000-0002-4152-1166
Somer L BishopDepartment of Psychiatry, University of California San Francisco, 401 Parnassus Ave., San Francisco, CA, 94143, USA.
Jane E RobertsDepartment of Psychology, University of South Carolina, 1512 Pendleton Street, Columbia, SC, 29208, USA.
University of South Carolina · USUniversity of California, San Francisco · US

Funding

Emergence, Stability and Predictors of Anxiety in Fragile X SyndromeR01MH107573 · NIMH · UNIVERSITY OF SOUTH CAROLINA AT COLUMBIA · PI Abigail Lee Hogan · 2016 to 2026
$6.1M
Supplement to The Emergence and Stability of Autism in Fragile X SyndromeR01MH090194 · NIMH · UNIVERSITY OF SOUTH CAROLINA AT COLUMBIA · PI ROBERTS, JANE E · 2011 to 2020
$5.4M
Development of a standardized measure of social-communication abilities for children with neurodevelopmental disordersR01HD093012 · NICHD · UNIVERSITY OF CALIFORNIA, SAN FRANCISCO · PI BISHOP, SOMER L. · 2017 to 2022
$3.2M
Early Features of Autism Risk in Down SyndromeF32HD097877 · NICHD · UNIVERSITY OF SOUTH CAROLINA AT COLUMBIA · PI WILL, ELIZABETH · 2018 to 2021
$175k
NICHD NIH HHS R01 HD093012NIMH NIH HHS L40 MH117727NIMH NIH HHS R01 MH090194NIMH NIH HHS R01 MH107573
6 · The paper itself

Abstract

backgroundAutism spectrum disorder (ASD) is highly prevalent in fragile X syndrome (FXS), affecting 50-70% of males. Motor impairments are a shared feature across autism and FXS that may help to better characterize autism in FXS. As motor skills provide a critical foundation for various language, cognitive, and social outcomes, they may serve an important mechanistic role for autism in FXS. As such, this study aimed to identify differences in motor trajectories across direct assessment and parent-report measures of fine and gross motor development between FXS with and without autism, and typical development, while controlling for cognitive functioning.

methodsThis prospective longitudinal study included 42 children with FXS, 24 of whom also had ASD (FXS + ASD), as well as 40 typically developing children. The Mullen Scales of Early Learning provided a direct measure of fine and gross motor skills, and the Vineland Adaptive Behavior Scales provided a measure of parent-reported fine and gross motor skills. Random slopes and random intercepts multilevel models were tested to determine divergence in developmental motor trajectories between groups when controlling for cognitive level.

resultsModel results indicated the children with FXS + ASD diverged from TD children by 9-months on all measures of gross and fine motor skills, even when controlling for cognitive level. Results also indicated an early divergence in motor trajectories of fine and gross motor skills between the FXS + ASD and FXS groups when controlling for cognitive level. This divergence was statistically significant by 18 months, with the FXS + ASD showing decelerated growth in motor skills across direct observation and parent-report measures.

conclusionsThis study is the first to examine longitudinal trends in motor development in children with FXS with and without comorbid ASD using both direct assessment and parent-report measures of fine and gross motor. Furthermore, it is among the first to account for nonverbal cognitive delays, a step towards elucidating the isolated role of motor impairments in FXS with and without ASD. Findings underscore the role of motor impairments as a possible signal representing greater underlying genetic liability, or as a potential catalyst or consequence, of co-occurring autism in FXS.

Indexed as

Autism Spectrum DisorderChild DevelopmentChild, PreschoolDevelopmental DisabilitiesFemaleFragile X SyndromeHumansInfantLongitudinal StudiesMaleMotor SkillsAutism spectrum disorderDevelopmental trajectoriesFragile X syndromeMotor development

Identifiers

PMID31586494
PMCPMC6778381
OpenAlexW2978018127

What OpenQuestion holds

Textmetadata
LicenceCC BY
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.