Social Attention and Theory of Mind in Preschool Aged Children with Sex Chromosome Trisomies (XXX, XXY, XYY): Predictive Value for Neurobehavioral Outcomes One Year Later

Background and objective: Sex Chromosome Trisomy (SCT) refers to having an extra X or Y chromosome (XXX, XXY, XYY), which occurs in approximately 1-2 of 1000 children. In this study, social attention, Theory of Mind (ToM) and neurobehavioral outcomes are investigated in preschool aged children with SCT compared to typically developing controls. Furthermore, the study aims to explore with a longitudinal design whether social attention and ToM in preschool aged children with SCT can be predictive of neurobehavioral outcomes one year later. Methods: In this study 71 children with SCT (aged 3-7) and 71 typically developing age matched controls participated. The Social Interaction Scanning (SIS) eye tracking paradigm was used to investigate social attention. ToM skills were measured with the ToM subtest of the Developmental NEuroPSYchological Assessment, second edition (NEPSY-II). Neurobehavioral outcomes were measured with the Social Responsiveness Scale, second edition (SRS-2),...Show moreBackground and objective: Sex Chromosome Trisomy (SCT) refers to having an extra X or Y chromosome (XXX, XXY, XYY), which occurs in approximately 1-2 of 1000 children. In this study, social attention, Theory of Mind (ToM) and neurobehavioral outcomes are investigated in preschool aged children with SCT compared to typically developing controls. Furthermore, the study aims to explore with a longitudinal design whether social attention and ToM in preschool aged children with SCT can be predictive of neurobehavioral outcomes one year later. Methods: In this study 71 children with SCT (aged 3-7) and 71 typically developing age matched controls participated. The Social Interaction Scanning (SIS) eye tracking paradigm was used to investigate social attention. ToM skills were measured with the ToM subtest of the Developmental NEuroPSYchological Assessment, second edition (NEPSY-II). Neurobehavioral outcomes were measured with the Social Responsiveness Scale, second edition (SRS-2), Strengths and Weaknesses of ADHD and Normal behavior (SWAN) and the Child Behavior Check List (CBCL) parental questionnaires respectively. Results: Results of the current study show a vulnerability for children with SCT in the domain of social attention to faces and eyes and ToM compared to typically developing controls. Findings are more pronounced for children aged 5-7 years old. Furthermore, children with SCT appear at increased risk for poorer neurobehavioral outcomes. Finally, results of the current study suggest that ToM may have a unique predictive value for social impairment over time, specifically in the domain of social communication and interaction. Secondly, social attention may have a unique predictive value for ADHD symptoms over time. Conclusions: Results indicate that screening of social cognitive functions (around the age of 5-7) and monitoring of neurodevelopmental pathways is important for clinical practice in SCT. In future research the efficacy of early tailored interventions targeting social cognitive skills in SCT can be investigated, to positively influence developmental outcomes of children with SCT.Show less

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Social Attention and Theory of Mind in Preschool Aged Children with Sex Chromosome Trisomies (XXX, XXY, XYY) Predictive Value for Neurobehavioral Outcomes One Year Later