Evidence map›Paper›PMID 39643599›Full record

ArticleAutism research : official journal of the International Society for Autism Research2025

Remote monitoring of social attention in neurogenetic syndromes and idiopathic neurodevelopmental disability.

Thomas W Frazier, Robyn M Busch, Patricia Klaas, Katherine Lachlan, Shafali Jeste, Alexander Kolevzon, Eva Loth, Jacqueline Harris, Tom Pepper, Kristin Anthony and 11 more

Abstract read
PubMed Full text at PMC Publisher
In one paragraph

Article in Autism research : official journal of the International Society for Autism Research, 2025. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 4 papers.

0numbers the graph read from it
0cells of the map it votes in
4citing 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

4 citing papers in PubMed.

  1. Research Review: Emotion regulation in individuals with intellectual disability - an integrative review.Journal of child psychology and psychiatry, and allied disciplines · 2026
    Review
  2. Article
  3. Remote monitoring of social attention in neurogenetic syndromes and idiopathic neurodevelopmental disability.Autism research : official journal of the International Society for Autism Research · 2025
    Article
  4. 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

21 authors.

Thomas W FrazierDepartment of Psychology, John Carroll University, University Heights, Ohio, USA.ORCID 0000-0002-6951-2667
Robyn M BuschDepartment of Neurology, Neurological Institute, Cleveland Clinic, Cleveland, Ohio, USA.
Patricia KlaasDepartment of Neurology, Neurological Institute, Cleveland Clinic, Cleveland, Ohio, USA.
Katherine LachlanHuman Genetics and Genomic Medicine, Faculty of Medicine, Southampton University and University Hospital Southampton NHS Foundation Trust, Southampton, UK.
Shafali JesteDivision of Neurology, Children's Hospital of Los Angeles, Los Angeles, California, USA.
Alexander KolevzonDepartments of Psychiatry and Pediatrics, Seaver Autism Center for Research and Treatment, Icahn School of Medicine at Mount Sinai, New York, New York, USA.
Eva LothDepartment of Forensic and Neurodevelopmental Science, Institute of Psychiatry, Psychology and Neuroscience Kings College London, London, UK.
Jacqueline HarrisDepartment of Neurology Kennedy, Krieger Institute and Johns Hopkins University School of Medicine, Baltimore, Maryland, USA.
Tom PepperPTEN Research Foundation, Cheltenham, UK.
Kristin AnthonyPTEN Hamartoma Tumor Syndrome Foundation, Huntsville, Alabama, USA.
J Michael GragliaSYNGAP Research Fund, San Diego, California, USA.
Kathryn HeldeSYNGAP Research Fund, San Diego, California, USA.
Christal DelagrammatikasMalan Syndrome Foundation, Old Bridge, New Jersey, USA.
Sandra Bedrosian-SermoneADNP Kids Research Foundation, Brush Prairie, Washington, USA.
Constance Smith-HicksDepartment of Neurology Kennedy, Krieger Institute and Johns Hopkins University School of Medicine, Baltimore, Maryland, USA.
Mustafa SahinRosamund Stone Zander Translational Neuroscience Center, Department of Neurology, Boston Children's Hospital and Harvard Medical School, Boston, Massachusetts, USA.
Eric A YoungstromInstitute for Mental and Behavioral Health Research and the Department of Psychiatry, Nationwide Children's Hospital and The Ohio State University, Columbus, Ohio, USA.
Charis EngGenomic Medicine Institute, Lerner Research Institute, Cleveland Clinic, Cleveland, Ohio, USA.
Lacey ChetcutiDepartment of Psychiatry and Behavioral Sciences, Stanford University, Stanford, California, USA.
Antonio Y HardanDepartment of Psychiatry and Behavioral Sciences, Stanford University, Stanford, California, USA.
Mirko UljarevicDepartment of Psychiatry and Behavioral Sciences, Stanford University, Stanford, California, USA.ORCID 0000-0002-7481-3923

Funding

The Impact of COVID-19 on People Living with Rare Diseases and Their FamiliesU2CTR002818 · NCATS · CINCINNATI CHILDRENS HOSP MED CTR · PI Maurizio Macaluso, Michael Wagner · 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
Genetic Analysis and Manipulation Core (GAEC)P50HD105351 · NICHD · BOSTON CHILDREN'S HOSPITAL · PI SCOTT Loren POMEROY, MUSTAFA SAHIN · 2021 to 2026
$9.4M
A Big Data Approach Toward the Development of New Quantitative Autism Severity Scores from Existing InstrumentsR15MH129815 · NIMH · JOHN CARROLL UNIVERSITY · PI FRAZIER, THOMAS WILLIAM · 2022 to 2022
$385k
ADNP Kids Research FoundationAutism Speaks 12776Malan Syndrome FoundationNCATS NIH HHS U2C TR002818NCATS NIH HHS U54NS092090NICHD NIH HHS P50 HD105351NIMH NIH HHS R15 MH129815NINDS NIH HHS U54 NS092090PTEN Research JCU-20-001RDCRN Data Management and Coordinating Center (DMCC) U2CTR002818Simons Foundation Autism Research Initiative 831500SynGAP Research Fund
6 · The paper itself

Abstract

Social attention is a key aspect of neurodevelopment and is significantly altered in neurodevelopmental genetic syndromes and many individuals with idiopathic autism spectrum disorder (ASD). The primary aim of the present study was to examine the psychometric properties of webcam-collected social attention measurements, including four new specific aspects of social attention, in three genetic syndromes (PTEN Hamartoma Tumor Syndrome-PHTS; Malan Syndrome-NFIX; and SYNGAP1-related disorder-SYNGAP1), a mixed group of other neurodevelopmental genetic syndromes (Other NDGS), and individuals with a range of idiopathic neurodevelopmental disorder (NDD). The secondary aim was to evaluate the construct validity of these social attention measurements, including evaluating known-groups validity across study groups and concurrent validity for separating ASD and non-ASD cases. Participants (N = 467, age 3-45; PHTS n = 102, NFIX n = 23, SYNGAP1 n = 42, other NDGS n = 63, idiopathic NDD n = 53, neurotypical siblings n = 71, and unrelated neurotypical controls n = 113) completed a 4-min online-administered social attention paradigm that includes a variety of distinct stimuli at three timepoints (baseline, 1-month, and 4-month follow-up). Social attention measures had good scale and test-retest reliability, with the exception of measures of non-social preference and face-specific processing. Unique patterns of social attention emerged across study groups, with near neurotypical levels in PHTS and weaker social attention in NFIX and SYNGAP1 relative to controls. Global social attention had good accuracy in detecting ASD within NDGS participants. Remote monitoring social attention, including distinct aspects of social attention, may be useful for characterizing phenotypic profiles and tracking the natural history of distinct NDGS and idiopathic NDD as well as identifying ASD within NDGS. Given their reproducibility and stability, global social attention and several distinct social attention measures may be useful outcomes for future clinical trials.

Indexed as

AttentionAutism Spectrum DisorderNeurodevelopmental DisordersSocial BehaviorAdolescentAdultChildChild, PreschoolFemaleHumansMaleMiddle AgedPsychometricsras GTPase-Activating ProteinsReproducibility of ResultsYoung Adultras GTPase-Activating ProteinsSYNGAP1 protein, humanautism spectrum disorderonlinereliabilityremote monitoringsocial attentionvalidity

Identifiers

PMID39643599
PMCPMC11826009

What OpenQuestion holds

Textmetadata
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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.