Challenges with shifting, regardless of disengagement: attention mechanisms and eye movements in Williams syndrome / Astrid HALLMAN in Journal of Neurodevelopmental Disorders, 17 (2025)  

Public ISBD [article]  inJournal of Neurodevelopmental Disorders > 17 (2025) Titre : Challenges with shifting, regardless of disengagement: attention mechanisms and eye movements in Williams syndrome Type de document : texte imprimé Auteurs : Astrid HALLMAN, Auteur ; Charlotte WILLFORS, Auteur ; Christine FAWCETT, Auteur ; Matilda A. FRICK, Auteur ; Ann NORDGREN, Auteur ; Johan Lundin KLEBERG, Auteur Langues : Anglais (eng) Mots-clés : Eye tracking  Intellectual disability  Orienting attention  Phasic alerting effect  Pupil dilation  Shifting attention  Visual disengagement  Williams syndrome  in accordance with the Declaration of Helsinki and received approval from the  Regional Ethics Committee of Stockholm, Sweden (dnr 2018/1218-31 with subsequent  amendments). Informed consents were collected from the participants and/or their  legal guardians. Informed assent was obtained from all participants who were able  to assent. Consent for publication: Not applicable. Competing interests: The  authors declare no competing interests. Index. décimale : PER Périodiques Résumé : BACKGROUND: People with Williams syndrome (WS) face challenges in various areas of cognitive processing, including attention. Previous studies suggest that these challenges are particularly pronounced when disengagement of attention from a previously attended stimulus is required, as compared to shifting attention without the need to disengage. Difficulties with attention could in turn be implicated in several of the behavioral characteristics of WS. Here, disengagement and shifting of visual attention, together with pupil dilation, were independently assessed in one of the largest eye-tracking studies of WS to date. METHODS: We investigated shifting, disengagement, and the effects of auditory alerting cues on pupil dilation in WS individuals (n = 45, age range = 9–58 years), non-WS individuals with intellectual disability (ID) (n = 36, age range = 6–59 years), and typically developed (TD) infants (n = 32, age range = 6–7 months), children and adults (n = 31, age range = 9–60 years), using a modified gap-overlap task. Data were analyzed using linear mixed-effect models (LMMs). RESULTS: Individuals with WS were less likely to shift their attention to upcoming targets than TD individuals (all ages), but more likely than the ID group to do so. When they did shift attention, participants with WS and ID were slower to initiate a gaze shift than TD participants regardless of whether disengagement was needed. In the WS group, failure to shift attention was strongly predicted by higher arousal (pupil dilation), which was induced by auditory alerting cues. CONCLUSIONS: Contrasting with previous theories of attention in WS, we found no evidence for a specific challenge in disengaging attention. Instead, our results point to a more general challenge in shifting attention. Reduced attention shifting in WS may be partly explained by atypical arousal regulation. These results contribute to our understanding of the WS phenotype. SUPPLEMENTARY INFORMATION: The online version contains supplementary material available at 10.1186/s11689-025-09639-z. En ligne : https://dx.doi.org/10.1186/s11689-025-09639-z Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=576 [article] Challenges with shifting, regardless of disengagement: attention mechanisms and eye movements in Williams syndrome [texte imprimé] / Astrid HALLMAN, Auteur ; Charlotte WILLFORS, Auteur ; Christine FAWCETT, Auteur ; Matilda A. FRICK, Auteur ; Ann NORDGREN, Auteur ; Johan Lundin KLEBERG, Auteur. Langues : Anglais (eng) in Journal of Neurodevelopmental Disorders > 17 (2025) Mots-clés : Eye tracking  Intellectual disability  Orienting attention  Phasic alerting effect  Pupil dilation  Shifting attention  Visual disengagement  Williams syndrome  in accordance with the Declaration of Helsinki and received approval from the  Regional Ethics Committee of Stockholm, Sweden (dnr 2018/1218-31 with subsequent  amendments). Informed consents were collected from the participants and/or their  legal guardians. Informed assent was obtained from all participants who were able  to assent. Consent for publication: Not applicable. Competing interests: The  authors declare no competing interests. Index. décimale : PER Périodiques Résumé : BACKGROUND: People with Williams syndrome (WS) face challenges in various areas of cognitive processing, including attention. Previous studies suggest that these challenges are particularly pronounced when disengagement of attention from a previously attended stimulus is required, as compared to shifting attention without the need to disengage. Difficulties with attention could in turn be implicated in several of the behavioral characteristics of WS. Here, disengagement and shifting of visual attention, together with pupil dilation, were independently assessed in one of the largest eye-tracking studies of WS to date. METHODS: We investigated shifting, disengagement, and the effects of auditory alerting cues on pupil dilation in WS individuals (n = 45, age range = 9–58 years), non-WS individuals with intellectual disability (ID) (n = 36, age range = 6–59 years), and typically developed (TD) infants (n = 32, age range = 6–7 months), children and adults (n = 31, age range = 9–60 years), using a modified gap-overlap task. Data were analyzed using linear mixed-effect models (LMMs). RESULTS: Individuals with WS were less likely to shift their attention to upcoming targets than TD individuals (all ages), but more likely than the ID group to do so. When they did shift attention, participants with WS and ID were slower to initiate a gaze shift than TD participants regardless of whether disengagement was needed. In the WS group, failure to shift attention was strongly predicted by higher arousal (pupil dilation), which was induced by auditory alerting cues. CONCLUSIONS: Contrasting with previous theories of attention in WS, we found no evidence for a specific challenge in disengaging attention. Instead, our results point to a more general challenge in shifting attention. Reduced attention shifting in WS may be partly explained by atypical arousal regulation. These results contribute to our understanding of the WS phenotype. SUPPLEMENTARY INFORMATION: The online version contains supplementary material available at 10.1186/s11689-025-09639-z. En ligne : https://dx.doi.org/10.1186/s11689-025-09639-z Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=576

Reversed gender ratio of autism spectrum disorder in Smith-Magenis syndrome / Heidi Elisabeth NAG in Molecular Autism, 9 (2018)  

Public ISBD [article]  inMolecular Autism > 9 (2018) . - 1p. Titre : Reversed gender ratio of autism spectrum disorder in Smith-Magenis syndrome Type de document : texte imprimé Auteurs : Heidi Elisabeth NAG, Auteur ; Ann NORDGREN, Auteur ; Britt-Marie ANDERLID, Auteur ; Terje NAERLAND, Auteur Article en page(s) : 1p. Langues : Anglais (eng) Mots-clés : Autism symptomatology  Gender  Smith-Magenis syndrome Index. décimale : PER Périodiques Résumé : Background: A substantial amount of research shows a higher rate of autistic type of problems in males compared to females. The 4:1 male to female ratio is one of the most consistent findings in autism spectrum disorder (ASD).Lately, the interest in studying ASD in genetic disorders has increased, and research has shown a higher prevalence of ASD in some genetic disorders than in the general population.Smith-Magenis syndrome (SMS) is a rare and complex genetic syndrome caused by an interstitial deletion of chromosome 17p11.2 or a mutation on the retinoic acid induced 1 gene. The disorder is characterised by intellectual disability, multiple congenital anomalies, obesity, neurobehavioural abnormalities and a disrupted circadian sleep-wake pattern. Methods: Parents of 28 persons with SMS between 5 and 50 years old participated in this study. A total of 12 of the persons with SMS were above the age of 18 at the time of the study. A total of 11 came from Sweden and 17 were from Norway.We collected information regarding the number of autism spectrum symptoms using the Social Communication Questionnaire (SCQ) and the Social Responsiveness Scale (SRS). Adaptive behaviour was also measured using the Vineland Adaptive Behavior Scale II. The level of intellectual disability was derived from a review of the medical chart. Results: We found significant gender differences in ASD symptomatology using the SCQ and SRS questionnaires. We found approximately three females per male above the SCQ cutoff. The same differences were not found in the intellectual level and adaptive behaviour or for behavioural and emotional problems.Gender had an independent contribution in a regression model predicting the total SCQ score, and neither the Vineland Adaptive Behavior Scale II nor the Developmental Behaviour Checklist had an independent contribution to the SCQ scores. Conclusion: We found a clear reversed gender difference in ASD symptomatology in persons with SMS. This may be relevant in the search for female protective factors assumed to explain the male bias in ASD. En ligne : http://dx.doi.org/10.1186/s13229-017-0184-2 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=354 [article] Reversed gender ratio of autism spectrum disorder in Smith-Magenis syndrome [texte imprimé] / Heidi Elisabeth NAG, Auteur ; Ann NORDGREN, Auteur ; Britt-Marie ANDERLID, Auteur ; Terje NAERLAND, Auteur . - 1p. Langues : Anglais (eng) in Molecular Autism > 9 (2018) . - 1p. Mots-clés : Autism symptomatology  Gender  Smith-Magenis syndrome Index. décimale : PER Périodiques Résumé : Background: A substantial amount of research shows a higher rate of autistic type of problems in males compared to females. The 4:1 male to female ratio is one of the most consistent findings in autism spectrum disorder (ASD).Lately, the interest in studying ASD in genetic disorders has increased, and research has shown a higher prevalence of ASD in some genetic disorders than in the general population.Smith-Magenis syndrome (SMS) is a rare and complex genetic syndrome caused by an interstitial deletion of chromosome 17p11.2 or a mutation on the retinoic acid induced 1 gene. The disorder is characterised by intellectual disability, multiple congenital anomalies, obesity, neurobehavioural abnormalities and a disrupted circadian sleep-wake pattern. Methods: Parents of 28 persons with SMS between 5 and 50 years old participated in this study. A total of 12 of the persons with SMS were above the age of 18 at the time of the study. A total of 11 came from Sweden and 17 were from Norway.We collected information regarding the number of autism spectrum symptoms using the Social Communication Questionnaire (SCQ) and the Social Responsiveness Scale (SRS). Adaptive behaviour was also measured using the Vineland Adaptive Behavior Scale II. The level of intellectual disability was derived from a review of the medical chart. Results: We found significant gender differences in ASD symptomatology using the SCQ and SRS questionnaires. We found approximately three females per male above the SCQ cutoff. The same differences were not found in the intellectual level and adaptive behaviour or for behavioural and emotional problems.Gender had an independent contribution in a regression model predicting the total SCQ score, and neither the Vineland Adaptive Behavior Scale II nor the Developmental Behaviour Checklist had an independent contribution to the SCQ scores. Conclusion: We found a clear reversed gender difference in ASD symptomatology in persons with SMS. This may be relevant in the search for female protective factors assumed to explain the male bias in ASD. En ligne : http://dx.doi.org/10.1186/s13229-017-0184-2 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=354

Williams syndrome: reduced orienting to other?s eyes in a hypersocial phenotype / Johan LUNDIN KLEBERG in Journal of Autism and Developmental Disorders, 53-7 (July 2023)  

Public ISBD [article]  inJournal of Autism and Developmental Disorders > 53-7 (July 2023) . - p.2786-2797 Titre : Williams syndrome: reduced orienting to other?s eyes in a hypersocial phenotype Type de document : texte imprimé Auteurs : Johan LUNDIN KLEBERG, Auteur ; Deborah M. RIBY, Auteur ; Christine FAWCETT, Auteur ; Hanna BJÖRLIN AVDIC, Auteur ; Matilda A. FRICK, Auteur ; Karin C. BROCKI, Auteur ; Jens HÖGSTRÖM, Auteur ; Eva SERLACHIUS, Auteur ; Ann NORDGREN, Auteur ; Charlotte WILLFORS, Auteur Article en page(s) : p.2786-2797 Langues : Anglais (eng) Index. décimale : PER Périodiques Résumé : Williams syndrome (WS) is a rare genetic condition associated with high sociability, intellectual disability, and social cognitive challenges. Attention to others' eyes is crucial for social understanding. Orienting to, and from other?s eyes was studied in WS (n 37, mean age 23, age range 9-53). The WS group was compared to a typically developing comparison participants (n 167) in stratified age groups from infancy to adulthood. Typically developing children and adults were quicker and more likely to orient to eyes than the mouth. This bias was absent in WS. The WS group had reduced peak saccadic velocities, indicating hypo-arousal. The current study indicates reduced orienting to others' eyes in WS, which may affect social interaction skills. En ligne : https://doi.org/10.1007/s10803-022-05563-6 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=508 [article] Williams syndrome: reduced orienting to other?s eyes in a hypersocial phenotype [texte imprimé] / Johan LUNDIN KLEBERG, Auteur ; Deborah M. RIBY, Auteur ; Christine FAWCETT, Auteur ; Hanna BJÖRLIN AVDIC, Auteur ; Matilda A. FRICK, Auteur ; Karin C. BROCKI, Auteur ; Jens HÖGSTRÖM, Auteur ; Eva SERLACHIUS, Auteur ; Ann NORDGREN, Auteur ; Charlotte WILLFORS, Auteur . - p.2786-2797. Langues : Anglais (eng) in Journal of Autism and Developmental Disorders > 53-7 (July 2023) . - p.2786-2797 Index. décimale : PER Périodiques Résumé : Williams syndrome (WS) is a rare genetic condition associated with high sociability, intellectual disability, and social cognitive challenges. Attention to others' eyes is crucial for social understanding. Orienting to, and from other?s eyes was studied in WS (n 37, mean age 23, age range 9-53). The WS group was compared to a typically developing comparison participants (n 167) in stratified age groups from infancy to adulthood. Typically developing children and adults were quicker and more likely to orient to eyes than the mouth. This bias was absent in WS. The WS group had reduced peak saccadic velocities, indicating hypo-arousal. The current study indicates reduced orienting to others' eyes in WS, which may affect social interaction skills. En ligne : https://doi.org/10.1007/s10803-022-05563-6 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=508

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