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1.
J Intellect Disabil Res ; 64(4): 296-302, 2020 04.
Article in English | MEDLINE | ID: mdl-32020687

ABSTRACT

BACKGROUND: Fragile X syndrome (FXS) is a single-gene disorder highly associated with anxiety; however, measuring anxiety symptoms in FXS and other neurogenetic syndromes is challenged by common limitations in language, self-awareness and cognitive skills required for many traditional assessment tasks. Prior studies have documented group-level differences in threat-related attentional biases, assessed via eye tracking, in FXS and non-FXS groups. The present study built on this work to test whether attentional biases correspond to clinical features of anxiety among adolescents and young adults with FXS. METHODS: Participants included 21 male adolescents with FXS ages 15-20 years who completed an adapted eye-tracking task that measured attentional bias towards fearful faces of varied emotional intensity. RESULTS: Among participants without anxiety disorders, attentional bias towards fear increased across age, similar to non-FXS paediatric anxiety samples. In contrast, participants with anxiety disorders exhibited greater stability in fear-related attentional biases across age. Across analyses, subtle fear stimuli were more sensitive to within-group anxiety variability than full-intensity stimuli. CONCLUSIONS: Our results provide novel evidence that although threat-related attentional biases may correspond with anxiety outcomes in FXS, these associations are complex and vary across developmental and task factors. Future studies are needed to characterise these associations in more robust longitudinal samples, informing whether and how eye-tracking tasks might be optimised to reliably predict and track anxiety in FXS.


Subject(s)
Anxiety Disorders/epidemiology , Anxiety Disorders/psychology , Attentional Bias/physiology , Fragile X Syndrome/epidemiology , Fragile X Syndrome/psychology , Adolescent , Adult , Anxiety Disorders/physiopathology , Comorbidity , Fragile X Syndrome/physiopathology , Humans , Male , Young Adult
2.
J Intellect Disabil Res ; 62(7): 625-636, 2018 07.
Article in English | MEDLINE | ID: mdl-29781139

ABSTRACT

BACKGROUND: Emerging evidence suggests that children with fragile X syndrome (FXS) exhibit abnormal gesture use early in development, although few studies have investigated the emergence of gesture use in this population or the impact of autism spectrum disorder (ASD) features on these behaviours. The present study examined the longitudinal development of gesture use in infants with FXS relative to low-risk controls and infant siblings of children with ASD (high-risk siblings), with the goal of establishing potentially unique patterns of gesture development in infants with FXS and understanding the relative impact of ASD symptom severity on these patterns. METHOD: Participants included 86 male infants (39 FXS, 27 high-risk siblings and 20 low-risk infants) assessed at 9, 12 and 24 months of age. Multilevel modelling was used to assess differences in number of gestures used and rates of gesture use across groups, as well as the relative impact of ASD symptom severity and nonverbal skills on these patterns. RESULTS: Infants with FXS used fewer gestures than high-risk siblings and low-risk infants, with this difference being primarily accounted for by the effect of low nonverbal abilities in the FXS group. Furthermore, although higher ASD symptom severity was associated with the use of fewer gestures in both the FXS and high-risk sibling groups, a significant amount of variance was shared between ASD symptom severity and nonverbal skills in FXS, but not in high-risk siblings. CONCLUSIONS: This study presents the first longitudinal analysis of early gesture development in FXS by using a multigroup design, clarifying the relative roles of cognitive deficits and ASD symptom severity in the development of gesture use in FXS. These findings offer novel evidence that early gesture use in FXS may reflect broader features of the FXS phenotype rather than predicting later social-communicative deficits characteristic of comorbid ASD.


Subject(s)
Autism Spectrum Disorder/complications , Autism Spectrum Disorder/physiopathology , Child Behavior/psychology , Fragile X Syndrome/complications , Fragile X Syndrome/physiopathology , Gestures , Autism Spectrum Disorder/psychology , Child, Preschool , Follow-Up Studies , Fragile X Syndrome/psychology , Humans , Infant , Longitudinal Studies , Male , Severity of Illness Index , Siblings/psychology
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