Evidence map›Paper›PMID 23200289›Full record

ArticleJournal of the American Academy of Child and Adolescent Psychiatry2012

Evidence of a distinct behavioral phenotype in young boys with fragile X syndrome and autism.

Jason J Wolff, James W Bodfish, Heather C Hazlett, Amy A Lightbody, Allan L Reiss, Joseph Piven

Abstract readComparative Study
In one paragraph

Article in Journal of the American Academy of Child and Adolescent Psychiatry, 2012. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 65 papers, 4 of them syntheses that pooled it.

0numbers the graph read from it
0cells of the map it votes in
65citing papers in PubMed, 4 pooled it
3.4field-weighted citation impact, top 7% 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

65 citing papers in PubMed, 4 syntheses or guidelines pooled it, 95 citations in OpenAlex.

  1. Pooled it
  2. Pooled it
  3. Pooled it
  4. Pooled it
  5. The impact of individual factors on linguistic alignment of autistic boys and their mothers.Autism : the international journal of research and practice · 2026
    Article
  6. Article
  7. Article
  8. Article
  9. Article
  10. Article
  11. Article
  12. Adaptive, behavioral, and cognitive outcomes in individuals with fragile X syndrome with varying autism severity.International journal of developmental neuroscience : the official journal of the International Society for Developmental Neuroscience · 2023
    Article
  13. Article
  14. Article
  15. Article
  16. Article
  17. Article
  18. Review
  19. Article
  20. Article

5 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

6 authors at 2 institutions in 1 country.

Jason J WolffCarolina Institute for Developmental Disabilities, University of North Carolina at Chapel Hill, NC 27599, USA. jason.wolff@cidd.unc.edu
James W Bodfish
Heather C Hazlett
Amy A Lightbody
Allan L Reiss
Joseph Piven
University of North Carolina at Chapel Hill · USStanford University · US

Funding

UNC MRDD Research CenterP30HD003110 · NICHD · UNIV OF NORTH CAROLINA CHAPEL HILL · PI BELGER, AYSENIL · 1985 to 2012
$28.7M
NEUROPSYCHIATRIC-MOLECULAR ASSOCIATIONS IN FRAGILE X SYNR01MH050047 · NIMH · STANFORD UNIVERSITY · PI REISS, ALLAN L · 1993 to 2022
$12.5M
Postdoctoral Research in Neurodevelopmental DisordersT32HD040127 · NICHD · UNIV OF NORTH CAROLINA CHAPEL HILL · PI BENJAMIN D PHILPOT, Mark D Shen · 2001 to 2026
$8.5M
Longitudinal MRI Study of Brain Development in Fragile XR01MH064708 · NIMH · STANFORD UNIVERSITY · PI PIVEN, JOSEPH, REISS, ALLAN L · 2002 to 2016
$8.1M
LONGITUDINAL MRI STUDY OF BRAIN DEVELOPMENT IN AUTISMR01MH061696 · NIMH · UNIVERSITY OF NORTH CAROLINA CHAPEL HILL · PI PIVEN, JOSEPH · 1999 to 2003
$1.9M
NICHD NIH HHS P30 HD003110NICHD NIH HHS P30HD03110NICHD NIH HHS T32 HD040127NIMH NIH HHS MH050047NIMH NIH HHS MH61696NIMH NIH HHS MH64708-05NIMH NIH HHS R01 MH050047NIMH NIH HHS R01 MH061696NIMH NIH HHS R01 MH064708
6 · The paper itself

Abstract

objectiveHow does the behavioral expression of autism in fragile X syndrome (FXS + Aut) compare with idiopathic autism (iAut)? Although social impairments and restricted, repetitive behaviors are common to these variants of autism, closer examination of these symptom domains may reveal meaningful similarities and differences. To this end, the specific behaviors comprising the social and repetitive behavioral domains in young children with FXS + Aut and iAut were profiled.

methodTwenty-three male subjects 3 to 5 years old with FXS + Aut were matched by age to a group of 38 boys with iAut. Repetitive behavior was assessed using the Repetitive Behavior Scales-Revised. Social behavior was evaluated using Autism Diagnostic Observation Schedule social item severity scores.

resultsRates of stereotypy, self-injury, and sameness behaviors did not differ between groups, whereas compulsive and ritual behavior scores were significantly lower for subjects with FXS + Aut compared with those with iAut. Those with FXS + Aut scored significantly lower (less severe) than the iAut group on five Autism Diagnostic Observation Schedule measurements of social behavior: gaze integration, quality of social overtures, social smile, facial expressions, and response to joint attention.

conclusionsThe behavioral phenotype of FXS + Aut and iAut are most similar with respect to lower-order (motoric) restricted, repetitive behaviors and social approach, but differ in more complex forms of restricted, repetitive behaviors and some social response behaviors. These findings highlight the phenotypic heterogeneity of autism overall and its unique presentation in an etiologically distinct condition.

Indexed as

Autistic DisorderFragile X SyndromeSocial BehaviorStereotyped BehaviorBehavioral SymptomsChild BehaviorChild, PreschoolDiagnosis, DifferentialDiagnostic and Statistical Manual of Mental DisordersFacial ExpressionFragile X Messenger Ribonucleoprotein 1HumansMaleMultivariate AnalysisPhenotypePsychiatric Status Rating ScalesFMR1 protein, humanFragile X Messenger Ribonucleoprotein 1

Identifiers

PMID23200289
PMCPMC3513689
OpenAlexW1978443173

What OpenQuestion holds

Textmetadata
LicenceTDM
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.