Abstract
BackgroundSex Chromosome Trisomies (SCTs; XXX, XXY, XYY) are genetic conditions that are associated with increased risk for neurodevelopmental problems and psychopathology. There is a great need for early preventive intervention programs to optimize outcome, especially considering the increase in prenatal diagnoses due to recent advances in non-invasive prenatal screening. This study is the first to evaluate efficacy of a neurocognitive training in children with SCT. As social behavioral problems have been identified as among the key areas of vulnerability, it was targeted at improving a core aspect of social cognition, the understanding of social cues from facial expressions.MethodsParticipants were 24 children with SCT and 18 typically developing children, aged 4–8 years old. Children with SCT were assigned to a training (n = 13) or waiting list (no-training) group (n = 11). Children in the training group completed a neurocognitive training program (The Transporters), aimed to increase understanding of facial emotions. Participants were tested before and after the training on facial emotion recognition and Theory of Mind abilities (NEPSY-II), and on social orienting (eyetracking paradigm). The SCT no-training group and typically developing control group were also assessed twice with the same time interval without any training. Feasibility of the training was evaluated with the Social Validity Questionnaire filled out by the parents and by children's ratings on a Visual Analog Scale.ResultsThe SCT training group improved significantly more than the SCT no-training and TD no-training group on facial emotion recognition (large effect size; = 0.28), performing comparable to typical controls after completing the training program. There were no training effects on ToM abilities and social orienting. Both children and parents expressed satisfaction with the feasibility of the training.ConclusionsThe significant improvement in facial emotion recognition, with large effect sizes, suggests that there are opportunities for positively supporting the development of social cognition in children with an extra X- or Y-chromosome, already at a very young age. This evidence based support is of great importance given the need for preventive and early training programs in children with SCT, aimed to minimize neurodevelopmental impact.
Highlights
Between 1:650 and 1:1000 children are born with a Sex Chromosome Trisomy [Sex Chromosome Trisomies (SCT); [1]]
We evaluated the efficacy of an emotion recognition training program in SCT on key areas of typically social cognitive development during early childhood that were found to be vulnerable in young children with SCT, i.e., on measures of emotion recognition, Theory of Mind and social orientation [14]
No difference between the SCT training group and the no-training group was found for receptive verbal ability
Summary
Between 1:650 and 1:1000 children are born with a Sex Chromosome Trisomy [SCT; [1]]. SCT is characterized by an extra X- or Y-chromosome compared to the typical karyotype of 46, XX in girls and 46, XY in boys. Intellectual functioning is typically within normal limits, somewhat lower on average, and SCT is related to a profile of specific cognitive vulnerabilities, for example in areas of executive functioning, language and social cognition [see for reviews: [2, 3]]. As SCT is a condition that are associated with increased risk for neurocognitive vulnerabilities and related neurobehavioral problems, these genetic conditions may serve as naturalistic “at risk” models of neurodevelopment. Sex Chromosome Trisomies (SCTs; XXX, XXY, XYY) are genetic conditions that are associated with increased risk for neurodevelopmental problems and psychopathology. As social behavioral problems have been identified as among the key areas of vulnerability, it was targeted at improving a core aspect of social cognition, the understanding of social cues from facial expressions. The total raw score range is between 1 and 35, with higher scores reflecting a better ability to recognize facial expressions
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