Evidence map›Paper›PMID 32406614›Full record

ArticleAutism research : official journal of the International Society for Autism Research2020

Psychometric Study of the Social Responsiveness Scale in Phelan-McDermid Syndrome.

Kellie Gergoudis, Alan Weinberg, Jonathan Templin, Cristan Farmer, Alison Durkin, Jordana Weissman, Paige Siper, Jennifer Foss-Feig, Maria Del Pilar Trelles, Jonathan A Bernstein and 8 more

Open access · greenAbstract read
In one paragraph

Article in Autism research : official journal of the International Society for Autism Research, 2020. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 15 papers, 1 of them a synthesis that pooled it.

0numbers the graph read from it
0cells of the map it votes in
15citing papers in PubMed, 1 pooled it
1.9field-weighted citation impact, top 14% 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

15 citing papers in PubMed, 1 synthesis or guideline pooled it, 27 citations in OpenAlex.

  1. Pooled it
  2. Trial
  3. Article
  4. Article
  5. Article
  6. Article
  7. Article
  8. Article
  9. Characterizing the autism spectrum phenotype in DYRK1A-related syndrome.Autism research : official journal of the International Society for Autism Research · 2023
    Article
  10. Updated consensus guidelines on the management of Phelan-McDermid syndrome.American journal of medical genetics. Part A · 2023
    Review
  11. Article
  12. Social behavioral impairments inFrontiers in pediatrics · 2023
    Article
  13. Review
  14. Article
  15. Observational
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

18 authors at 7 institutions in 2 countries.

Kellie GergoudisIcahn School of Medicine at Mount Sinai, New York, New York, USA.ORCID 0000-0003-2393-8563
Alan WeinbergDepartment of Population Health Science and Policy, Icahn School of Medicine at Mount Sinai, New York, New York, USA.
Jonathan TemplinDepartment of Psychology and Research in Education, University of Kansas, Lawrence, Kansas, USA.
Cristan FarmerPediatrics and Developmental Neuroscience Branch, National Institute of Mental Health, National Institutes of Health, Bethesda, Maryland, USA.
Alison DurkinSeaver Autism Center, Department of Psychiatry, Icahn School of Medicine at Mount Sinai, New York, New York, USA.
Jordana WeissmanSeaver Autism Center, Department of Psychiatry, Icahn School of Medicine at Mount Sinai, New York, New York, USA.
Paige SiperSeaver Autism Center, Department of Psychiatry, Icahn School of Medicine at Mount Sinai, New York, New York, USA.
Jennifer Foss-FeigSeaver Autism Center, Department of Psychiatry, Icahn School of Medicine at Mount Sinai, New York, New York, USA.ORCID 0000-0002-1491-8248
Maria Del Pilar TrellesSeaver Autism Center, Department of Psychiatry, Icahn School of Medicine at Mount Sinai, New York, New York, USA.
Jonathan A BernsteinDepartment of Pediatrics, Stanford University School of Medicine, Stanford, California, USA.ORCID 0000-0001-5369-346X
Joseph D BuxbaumSeaver Autism Center, Department of Psychiatry, Icahn School of Medicine at Mount Sinai, New York, New York, USA.
Elizabeth Berry-KravisDepartment of Pediatrics, Rush University Medical Center, Chicago, Illinois, USA.
Craig M PowellDepartment of Neurobiology, University of Alabama at Birmingham, Birmingham, Alabama, USA.ORCID 0000-0001-5451-2165
Mustafa SahinF.M. Kirby Neurobiology Center, Boston Children's Hospital, Harvard Medical School, Boston, Massachusetts, USA.
Latha SooryaDepartment of Psychiatry, Rush University Medical Center, Chicago, Illinois, USA.ORCID 0000-0002-0414-4533
Audrey ThurmPediatrics and Developmental Neuroscience Branch, National Institute of Mental Health, National Institutes of Health, Bethesda, Maryland, USA.ORCID 0000-0002-0489-9485
Alexander KolevzonSeaver Autism Center, Department of Psychiatry, Icahn School of Medicine at Mount Sinai, New York, New York, USA.
Developmental Synaptopathies ConsortiumDepartment of Neurology, Boston Children's Hospital, Harvard Medical School, Boston, Massachusetts, USA.
Icahn School of Medicine at Mount Sinai · USNational Institutes of Health · USRush University Medical Center · USBoston Children's Hospital · USCivitan International · USStanford University · USUniversity of Kansas · US

Funding

The Impact of COVID-19 on People Living with Rare Diseases and Their FamiliesU2CTR002818 · NCATS · CINCINNATI CHILDRENS HOSP MED CTR · PI Maurizio Macaluso · 2019 to 2026
$51.2M
Toward better characterization and clinical trial readiness for targeting neuropsychiatric manifestations in PTEN pathogenic variantsU54NS092090 · NINDS · BOSTON CHILDREN'S HOSPITAL · PI M. Pilar Trelles · 2014 to 2026
$20.1M
Neurodevelopmental and Behavioral Phenotyping ZICMH002961 · NIMH · NATIONAL INSTITUTE OF MENTAL HEALTH · PI THURM, AUDREY · 2017 to 2025
$12.6M
NOVEL GENETIC MODELS OF AUTISMR01HD069560 · NICHD · UT SOUTHWESTERN MEDICAL CENTER · PI POWELL, CRAIG M · 2011 to 2020
$4.8M
Electrophysiological Markers for Interventions in Phelan-McDermid Syndrome and Idiopathic AutismR01NS105845 · NINDS · ICAHN SCHOOL OF MEDICINE AT MOUNT SINAI · PI KOLEVZON, ALEXANDER · 2018 to 2022
$3.7M
Molecular and Cellular Basis of Neurodevelopmental DisordersR01MH113948 · NIMH · UNIVERSITY OF ALABAMA AT BIRMINGHAM · PI POWELL, CRAIG M · 2020 to 2024
$3.3M
Intramural NIH HHS ZIC MH002961NCATS NIH HHS U2C TR002818NICHD NIH HHS R01 HD069560NIMH NIH HHS R01 MH113948NIMH NIH HHS ZICMH002961NINDS NIH HHS R01 NS105845NINDS NIH HHS U54 NS092090NINDS NIH HHS U54NS092090
6 · The paper itself

Abstract

The Social Responsiveness Scale-2 (SRS-2) is a quantitative measure used to characterize symptoms of autism spectrum disorder (ASD). However, research suggests that SRS-2 scores are significantly influenced by language ability and intellectual disability (ID). Efforts to refine the SRS-2 by Sturm, Kuhfeld, Kasari, and Mccracken [Journal of Child Psychology and Psychiatry, 58(9), 1053-1061] yielded a shortened form, yet its psychometric properties in populations with severe ID remain unknown. This study aims to examine the psychometric properties of the SRS-2 in Phelan-McDermid syndrome (PMS), a genetic condition associated with ASD and ID, thereby guiding score interpretation in this population and future development of targeted scales. Analyses, including Item Response Theory (IRT), were conducted on a sample of individuals with PMS (n = 91) recruited at six sites nationally. Psychometric properties evaluated include measures of reliability (internal consistency, test-retest reliability) and validity (structural, construct, content). While both SRS-2 forms are reliable, the shortened SRS-2 shows superior validity to the full SRS-2 for measuring ASD symptoms in PMS. On IRT analysis, the shortened SRS-2 shows excellent discrimination and precisely evaluates respondents across a wide range of ASD symptomatology but interpretation is limited by uncertain content validity and small sample size. The shortened SRS-2 shows some promise for use in PMS, but future refinements and additions are needed to develop items that are tailored to identify ASD in children with severe ID and specifically PMS. LAY SUMMARY: This study determined that a shortened form of the Social Responsiveness Scale, Second Edition (SRS-2) shows both promise and limitations for the characterization of autism symptomatology in individuals with Phelan-McDermid syndrome (PMS), a population characterized by intellectual disability (ID). Caution should be used when interpreting SRS-2 scores in individuals with ID and future research should modify existing items and develop new items to improve the SRS-2's ability to accurately characterize autism symptomatology in PMS. Autism Res 2020, 13: 1383-1396. © 2020 International Society for Autism Research, Wiley Periodicals, Inc.

Indexed as

AdolescentAutism Spectrum DisorderBehavior Rating ScaleChildChild, PreschoolChromosome DeletionChromosome DisordersChromosomes, Human, Pair 22FemaleHumansIntellectual DisabilityMalePsychometricsReproducibility of ResultsSocial SkillsYoung Adultautism spectrum disorderbehavioral measuresintellectual disabilityItem Response TheoryPhelan-McDermid syndrome

Identifiers

PMID32406614
PMCPMC8103889
OpenAlexW3025767169

What OpenQuestion holds

Textmetadata
LicenceTDM
Read underepoch 390

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.