Seks Chromosoom Trisomie (SCT) is een chromosomale afwijking waarbij kinderen worden geboren met een extra X- of Y-chromosoom. Kinderen met SCT lopen een hoger risico op problemen in sociaal...Show moreSeks Chromosoom Trisomie (SCT) is een chromosomale afwijking waarbij kinderen worden geboren met een extra X- of Y-chromosoom. Kinderen met SCT lopen een hoger risico op problemen in sociaal functioneren en op het ontwikkelen van een Autisme Spectrum Stoornis (ASS). Onderzoek naar sociaal functioneren en de relatie met ASS bij kinderen met SCT is beperkt. Empathie wordt beschouwd als belangrijk voor sociaal functioneren en lijkt verminderd te zijn bij kinderen met SCT. Deze studie heeft als doel te onderzoeken of de relatie tussen empathie, sociale gedragsvaardigheden en autisme-eigenschappen verschilt voor kinderen met SCT en controles. Deelnemers aan dit onderzoek waren 37 kinderen met SCT en 38 controles. Kinderen werden beoordeeld tijdens een test van 120 minuten. Ouders werd gevraagd om vragenlijsten in te vullen terwijl het kind werd getest. Om sociale gedragsvaardigheden te meten (Gedeelde Aandacht, Sociale Interactie en Gedragsverzoeken) werd de ESCS gebruikt. Om empathie te meten werd de EmQue-vragenlijst gebruikt, en om autisme-eigenschappen te meten werd de SRS-2 gebruikt. Kinderen met SCT vertoonden significant meer autisme-eigenschappen dan controles. Kinderen met SCT toonden ook significant minder empathie dan controles. Kinderen met SCT verschilden niet van controles in Gedeelde Aandacht of Sociale Interactie. Ze verschilden wel van controles in Gedragsverzoeken. Empathie en sociale gedragsvaardigheden verklaarden niet het verschil in autisme-eigenschappen tussen kinderen met SCT en controles. De interactie tussen de leeftijd van de kinderen en de onderzoeksgroep bleek net niet significant in het verklaren van het verschil in autisme-eigenschappen tussen SCT en controles.Show less
Children with Klinefelter may face difficulties in their later life when it comes to social emotional development and behavior problems (Bruining et al., 2009). This could lead to behavior- and...Show moreChildren with Klinefelter may face difficulties in their later life when it comes to social emotional development and behavior problems (Bruining et al., 2009). This could lead to behavior- and emotion regulation problems including anxiety. During a stressful life event it is crucial to be able to control/use cognitive emotion regulation strategies (Thompson, 1994). If this fails, it can lead to behavior- or social development problems. Although separate results from previous studies suggest a link between anxiety, regulation problems and stressful life events, this possible coherence has not been studied yet for Klinefelter children. Method: To investigate this, 95 children in the age of 1-7 years (of which 51 children with Klinefelter) and their parents participated in a series of questionnaires (Child Behavior Checklist, Children’s Behavior-/Early Childhood Behavior-/VMG Life Events Questionnaire). Results: Children with Klinefelter were found to be more anxious and showed a less expressive internal regulation state compared to the control group, according to their parents. According to the CBQ/ECBQ Reactivity/Soothability subscale results, children with Klinefelter showed less expressive (blunted) emotions when they were in pain, upset or experienced an exciting activity/event. In addition, stressful life events turned out to be a predictor for anxious depressed behavior in children with Klinefelter. There was also no interaction found for the degree of stressful life events and anxious/depressed or reactivity/soothability behavior within the Klinefelter- or control group. Conclusion: Children with Klinefelter experience more anxiety/depressed behavior and less expressive internal regulation state than children without Klinefelter. An association between stressful life events and anxiety/depressed behavior is found within children with Klinefelter, but there is no evidence found for a coherence between stressful life events and regulation problems. Furthermore, no interaction was found between the degree of stressful life events and anxious/depressed or reactivity/soothability behavior within the Klinefelter- or control group.Show less
Research master thesis | Developmental Psychopathology in Education and Child Studies (research) (MSc)
closed access
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...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
This research describes the correlation and predictability of social functioning and cognitive functioning of men with Klinefelter’s syndrome. In this research, data from an ongoing study by the...Show moreThis research describes the correlation and predictability of social functioning and cognitive functioning of men with Klinefelter’s syndrome. In this research, data from an ongoing study by the University of Leiden was used and combined with new data that was recruited. The study was performed on 64 men, ranging in age between 16 and 57 years old (M = 37.98, SD = 11.22). Several t-tests for independent samples, MANOVA’s, Pearson’s r correlations coefficients and a multiple regression was used. The results show more impairments in social functioning for men with Klinefelter’s syndrome than for the control group. With regard to the cognitive functions, the results show no significant difference for sustained attention between the Klinefelter group and the control group. The quality of language was different for the Klinefelter group and results show a correlation for social functioning within the Klinefelter group, which means the more restrictions there are in social functioning, the lower the quality of language. The quality of language was however not a significant predictor for the social functioning of men with Klinefelter. Also the social-cognitive functioning was significantly different between the Klinefelter group and the control group, however no correlations were found. Due to a lack of literature about cognitive functioning and social functioning of men with Klinefelter’s syndrome, more research is needed on this subject. In future studies there is also more research needed about the correlation between quality of language and social functioning, so more is known about the impairments and needs of men with Klinefelter’s syndrome. Results from future studies can be used to adjust the interventions, and probably early interventions, to the needs of men with Klinefelter’s syndrome.Show less
In dit onderzoek is onderzocht of kinderen met een extra x-chromosoom kwetsbaarder zijn voor de ontwikkeling van schizotypische kenmerken en daarmee een mogelijk verhoogd risico hebben op...Show moreIn dit onderzoek is onderzocht of kinderen met een extra x-chromosoom kwetsbaarder zijn voor de ontwikkeling van schizotypische kenmerken en daarmee een mogelijk verhoogd risico hebben op psychotische stoornissen. Bij 150 kinderen tussen de 7 en 20 jaar zijn de Kiddie Formal Thought Disorder Story game (kFTDS) en de Schizotypal Personality Questionnaire (SPQ-k) afgenomen. De steekproef bestaat uit 95 controlekinderen, 32 jongens met Klinefelter en 26 meisjes met Triple-X. De resultaten wijzen uit dat de klinische groep kinderen met een extra x-chromosoom meer schizotypische kenmerken en denkfouten laat zien dan de controlekinderen. Er kan dus geconcludeerd worden dat zij op kinderleeftijd al meer moeite hebben met de regulatie van hun denken dan controlekinderen, waardoor het belangrijk is dat er vroegtijdig gelet wordt op voorlopers van psychoses. Daarnaast zijn de resultaten een indicatie voor de rol van het extra x-chromosoom in de kwetsbaarheid voor de ontwikkeling van een psychose.Show less
Research master thesis | Developmental Psychopathology in Education and Child Studies (research) (MSc)
open access
2017-09-20T00:00:00Z
One of the dysfunctions associated with Klinefelter syndrome (KS) (47,XXY) are difficulties in social functioning, which may arise as a consequence of executive functioning (EF) and Theory of Mind ...Show moreOne of the dysfunctions associated with Klinefelter syndrome (KS) (47,XXY) are difficulties in social functioning, which may arise as a consequence of executive functioning (EF) and Theory of Mind (ToM) deficits. In this study, the influence of EF on ToM skills in KS boys was examined, as well as how these skills relate to autistic features. ASD traits were assessed with the parent-report Autism-Spectrum Quotient. ToM skills were measured with the Social Cognitive Skills Test. EF skills were measured with the Clinical Evaluation of Language Fundamentals and Amsterdam Neuropsychological Tasks. 28 KS boys (Mage=13.2, SD=3.0) participated in this study, along with 45 ASD boys (Mage=11.9, SD=2.0) and 46 boys from the general population (Mage=12.2, SD=2.9) as control groups. KS boys showed substantially elevated levels of ASD traits compared to the general population, but lower levels than ASD boys. In addition, difficulties regarding ToM and cognitive flexibility were more prominent in KS boys than in ASD boys compared to the general population. ToM task performance could partly be explained by spatial WM and attention switching (as autistic trait) could partly be explained by ToM skills in KS boys. The current study highlights the importance of attention modulation skills and ToM skills in social functioning of individuals with KS, providing promising insights regarding prevention and intervention. Future studies should focus on the effectiveness of enhancing EF skills in KS boys and the role of the X chromosome in vulnerability for autistic traits.Show less