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

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.

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Journal
Autism Research
Published
2024-12-06
DOI
https://doi.org/10.1002/aur.3290
Citations
1
Primary Topic
Autism Spectrum Disorder Research
Type
article
Field-Weighted Citation Impact
0.34

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article

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

Constance L. Smith‐Hicks, Alexander Kolevzon, Lacey Chetcuti, Sandra Bedrosian‐Sermone et al.
1 citations
Autism Research
Autism Spectrum Disorder Research
0.34
article

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

Constance L. Smith‐Hicks, Alexander Kolevzon, Lacey Chetcuti, Sandra Bedrosian‐Sermone, Eric A. Youngstrom, Kristin Anthony, Charis Eng, J. Michael Graglia, Patricia Klaas, Robyn M. Busch, Antonio Y. Hardan, Thomas William Frazier, Jacqueline K. Harris, Mirko Uljarević, Christal G. Delagrammatikas, Katherine Lachlan, Thomas Pepper, Mustafa Şahin, Shafali Jeste, Eva Loth, Kathryn A. Helde
article en
1 citations

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.

Autism ResearchVol. 18(2)
Boston Children's Hospital (US), Kennedy Krieger Institute (US), Nationwide Children's Hospital (US), Cleveland Clinic (US), Children's Hospital of Los Angeles (US), Harvard University (US), Johns Hopkins University (US), John Carroll University (US), King's College London (GB), SUNY Upstate Medical University (US), University Hospital Southampton NHS Foundation Trust (GB), Cleveland Clinic Lerner College of Medicine (US), Rome Foundation (US), PTEN Hamartoma Tumor Syndrome Foundation (US), PTEN Research Foundation (GB), Center for Pain and the Brain (US), The Ohio State University (US), Stanford University (US), Icahn School of Medicine at Mount Sinai (US)
Autism Speaks, Bristol-Myers Squibb, Roche, Biogen, F. Hoffmann-La Roche, GW Pharmaceuticals, Simons Foundation Autism Research Initiative, PTEN Research, Rare Diseases Clinical Research Network, Quadrant Biosciences, National Institutes of Health, National Center for Advancing Translational Sciences
Reduced inequalities
Openalex Percentile: Top 38%
Autism Spectrum Disorder Research
0.34
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