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Nienke Bouw

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Armando Marques-Guedes

UNL - New University of Lisbon

Daniel Deslauriers

California Institute of Integral Studies

Macquarie University

Universidade Federal do Rio Grande do Sul

Abant Izzet Baysal University, Bolu, Turkey

Prof. Dr. Raffaele Pisano, MSc, HDR (Habil.)

Université des Sciences et Technologies de Lille (Lille-1)

Universität Heidelberg

Budapest University of Technology and Economics

Patrick Blessinger

St. John's University

Università degli Studi di Padova

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Papers by Nienke Bouw

The Impact of Sex Chromosome Trisomies (XXX, XXY, XYY) on Social Attention and Affect Recognition: A Cross-Sectional Eye Tracking Study

Research Square (Research Square), Jun 21, 2021

Background About 1:650-1000 children are born with an extra X or Y chromosome (47,XXX; 47,XXY; 47... more Background About 1:650-1000 children are born with an extra X or Y chromosome (47,XXX; 47,XXY; 47,XYY), which results in a Sex Chromosome Trisomy (SCT). This international cross-sectional study was designed to investigate social attention and affect recognition during early life of children with SCT, with the aim to nd indicators for support and treatment. Methods A group of 101 children with SCT (aged 1-7 years old; M age = 3.7 years) was included in this study, as well as a population-based sample of 98 children without SCT (M age = 3.7). Social attention was measured using an eye tracking method that quanti es rst xations and xation durations on eyes of static faces and xation durations on social information (eyes, faces) in a dynamic paradigm (with two conditions: single face and multiple faces). Affect recognition was measured using the subtest Affect Recognition of the NEPSY-II neuropsychological test battery. Recruitment and assessment took place in the Netherlands and the United States. Results Eyetracking results reveal that children with SCT show lower proportions xation duration on social information already from the age of three years, compared to children without SCT. Also, impairments in the clinical range for affect recognition were found (32.2% of the SCT group scored in the well below average range); these di culties increase with age. Conclusions These results highlight the importance to further explore the development of social cognition skills of children with SCT in a longitudinal design, the monitoring of affect recognition skills, and the implementation of (preventive) interventions aiming to support the development of social attention and affect recognition.

Early impact of X‐ and Y‐chromosome variations ( <scp>XXX</scp> , <scp>XXY</scp> , <scp>XYY</scp> ) on social communication and social emotional development in 1–2‐year‐old children

American Journal of Medical Genetics, Mar 14, 2022

Sex chromosome trisomies (SCTs) are characterized by an extra X-or Y-chromosome (XXX, XXY, XYY). ... more Sex chromosome trisomies (SCTs) are characterized by an extra X-or Y-chromosome (XXX, XXY, XYY). The present study aims to investigate early signs of social communication and social emotional development in very young children with SCT. Thirty-four children with SCT (aged 12-24 months) were included in this study, as well as 31 agematched controls. Social communication was measured with structured behavior observations according to the Early Social Communication Scales, and social emotional developmental level with the Bayley Social Emotional parental questionnaire. Recruitment and assessment took place in the Netherlands and in the United States. On average, 12-24-month old children with SCT showed difficulties with early social communication, more so in responding to others as compared to initiating social communications. During social interactions, children with SCT made less frequent eye contact, compared to controls. Also, difficulties in acquiring social emotional milestones were found in 1-year old children with SCT, with 44% of the children having social emotional vulnerabilities in the borderline or extremely low range, compared to typically developing children. In this cohort, no significant predictive effects of karyotype-subtype (XXX, XXY, XYY) were found. Already from a very early age, SCT can be associated with increased risk for vulnerabilities in adaptive social functioning. These findings suggest that SCT impact the maturation of the social brain already from an early age, and stress the importance of early monitoring and (preventive) support early social development in young children with SCT.

The impact of sex chromosome trisomies (XXX, XXY, XYY) on gaze towards faces and affect recognition: a cross-sectional eye tracking study

Journal of Neurodevelopmental Disorders, Aug 2, 2022

Background: About 1:650-1000 children are born with an extra X or Y chromosome (47,XXX; 47,XXY; 4... more Background: About 1:650-1000 children are born with an extra X or Y chromosome (47,XXX; 47,XXY; 47,XYY), which results in a sex chromosome trisomy (SCT). This international cross-sectional study was designed to investigate gaze towards faces and affect recognition during early life of children with SCT, with the aim to find indicators for support and treatment. Methods: A group of 101 children with SCT (aged 1-7 years old; M age = 3.7 years) was included in this study, as well as a population-based sample of 98 children without SCT (M age = 3.7). Eye gaze patterns to faces were measured using an eye tracking method that quantifies first fixations and fixation durations on eyes of static faces and fixation durations on eyes and faces in a dynamic paradigm (with two conditions: single face and multiple faces). Affect recognition was measured using the subtest Affect Recognition of the NEPSY-II neuropsychological test battery. Recruitment and assessment took place in the Netherlands and the USA. Results: Eye tracking results reveal that children with SCT show lower proportion fixation duration on faces already from the age of 3 years, compared to children without SCT. Also, impairments in the clinical range for affect recognition were found (32.2% of the SCT group scored in the well below average range). Conclusions: These results highlight the importance to further explore the development of social cognitive skills of children with SCT in a longitudinal design, the monitoring of affect recognition skills, and the implementation of (preventive) interventions aiming to support the development of attention to social important information and affect recognition.

Early Social Behavior in Young Children with Sex Chromosome Trisomies (XXX, XXY, XYY): Profiles of Observed Social Interactions and Social Impairments Associated with Autism Spectrum Disorder (ASD)

Journal of Autism and Developmental Disorders, May 12, 2022

Individuals with Sex Chromosome Trisomies (SCT; XXX, XXY, XYY) have an increased vulnerability fo... more Individuals with Sex Chromosome Trisomies (SCT; XXX, XXY, XYY) have an increased vulnerability for developing challenges in social adaptive functioning. The present study investigates social interaction behavior in the context of varying social load, and Autism Spectrum Disorder (ASD) symptomatology in young children aged 1-7.5 years old, with SCT (N = 105) and control children (N = 101). Children with SCT show less interaction behaviors and more social withdrawal, as compared to their control peers, which were most evident in the high social load condition. Second, social impairments related to ASD are more prevalent, as compared to controls (27.1% at clinical level). These findings stress the importance of early monitoring and (preventive) support of early social development in young children with SCT.

Early symptoms of autism spectrum disorder (ASD) in 1–8 year old children with sex chromosome trisomies (XXX, XXY, XYY), and the predictive value of joint attention

European Child & Adolescent Psychiatry, Sep 15, 2022

The objective of the present study is to investigate the impact of Sex Chromosome Trisomy (SCT; X... more The objective of the present study is to investigate the impact of Sex Chromosome Trisomy (SCT; XXX, XXY, XYY) on the early appearance of Autism Spectrum Disorder (ASD) symptoms, and the predictive value of Joint Attention for symptoms of ASD. SCTs are specific genetic conditions that may serve as naturalistic 'at risk' models of neurodevelopment, as they are associated with increased risk for neurobehavioral vulnerabilities. A group of 82 children with SCT (aged 1-8 years) was included at baseline of this longitudinal study. Joint Attention was measured at baseline with structured behavior observations according to the Early Social Communication Scales. ASD symptoms were assessed with the Modified Checklist for Autism in Toddlers questionnaire and Autism Diagnostic Interview-Revised in a 1-year follow-up. Recruitment and assessment took place in the Netherlands and in the United States. The results demonstrate that ASD symptoms were substantially higher in children with SCT compared to the general population, with 22% of our cohort at clinical risk for ASD, especially in the domain of social interaction and communication. Second, a predictive value of Joint Attention was found for ASD symptoms at 1-year follow-up. In this cohort, no differences were found between karyotype-subtypes. In conclusion, from a very early age, SCT can be associated with an increased risk for vulnerabilities in adaptive social functioning. These findings show a neurodevelopmental impact of the extra X or Y chromosome on social adaptive development associated with risk for ASD already from early childhood onward. These findings advocate for close monitoring and early (preventive) support, aimed to optimize social development of young children with SCT.

The Impact of Sex Chromosome Trisomies (XXX, XXY, XYY) on Early Social Cognition: Social Orienting, Joint Attention, and Theory of Mind

Archives of Clinical Neuropsychology, 2021

Objective About 1:650–1,000 children are born with an extra X or Y chromosome (XXX; XXY; XYY), wh... more Objective About 1:650–1,000 children are born with an extra X or Y chromosome (XXX; XXY; XYY), which results in a sex chromosome trisomy (SCT). This study aims to cross-sectionally investigate the impact of SCT on early social cognitive skills. Basic orienting toward social cues, joint attention, and theory of mind (ToM) in young children with SCT were evaluated. Method About 105 children with SCT (range: 1–7 years old) were included in this study, as well as 96 age-matched nonclinical controls. Eyetracking paradigms were used to investigate the eye gaze patterns indicative of joint attention skills and orienting to social interactions. The ToM abilities were measured using the subtest ToM of the Developmental NEuroPSYchological Assessment, second edition, neuropsychological test battery. Recruitment and assessment took place in the Netherlands and in the United States. Results Eyetracking results revealed difficulties in children with SCT in social orienting. These difficulties wer...

Mindfulness-Based Stress Reduction in Prison: Experiences of Inmates, Instructors, and Prison Staff

International Journal of Offender Therapy and Comparative Criminology, 2019

Mindfulness intervention aims to reduce stress and to improve physical and mental health. The pre... more Mindfulness intervention aims to reduce stress and to improve physical and mental health. The present study investigated feasibility and effectiveness of mindfulness intervention in a prison context, in both a qualitative and quantitative fashion. Specifically, the mindfulness-based stress reduction (MBSR) intervention was investigated, in a retrospective pre–post design, in five Dutch prisons. Twenty-two inmates (out of 25 approached, mean age: 40.1 years ( SD = 11.1), convicted of murder, manslaughter, sexual offenses, drug offenses, robbery with violence, and/or illegal restraint/kidnap, and sentenced to incarceration between 15 and 209 months ( M = 5.5 years; SD = 3.8) took part in a semistructured interview after completion of the MBSR intervention. The interviews addressed level of satisfaction and challenges regarding the MBSR intervention as well as potential effects on stress responsivity, coping style, impulse control, aggression, and self-esteem. Ten staff members and fou...

Empathic distress and concern predict aggression in toddlerhood: The moderating role of sex

Infant Behavior and Development, 2019

This is an open access article under the terms of the Creat ive Commo ns Attri bution-NonCo mmerc... more

Neurocognitive and behavioral development in young children (1-7 years) with sex chromosome trisomy

Endocrine connections, Mar 1, 2023

Investigating sex chromosome trisomies (SCTs) may help in understanding neurodevelopmental pathwa... more Investigating sex chromosome trisomies (SCTs) may help in understanding neurodevelopmental pathways underlying the risk for neurobehavioral problems and psychopathology. Knowledge about the neurobehavioral phenotype is needed to improve clinical care and early intervention for children with SCT. This is especially relevant considering the increasing number of early diagnosed children with the recent introduction of noninvasive prenatal screening. The TRIXY Early Childhood Study is a longitudinal study designed to identify early neurodevelopmental risks in children with SCT, aged 1-7 years. This review summarizes the results from the TRIXY Early Childhood Study, focusing on early behavioral symptoms in areas of autism spectrum disorder, attention-deficit hyperactivity disorder, and communication disorders, and underlying neurocognitive mechanisms in domains of language, emotion regulation, executive functioning, and social cognition. Behavioral symptoms were assessed through structured behavior observation and parental questionnaires. Neurocognition was measured using performance tests, eyetracking, and psychophysiological measures of arousal. In total, 209 children aged 1-7 years were included: 107 children with SCT (33 XXX, 50 XXY, and 24 XYY) and 102 age-matched population controls. Study outcomes showed early behavioral symptoms in young children with SCT, and neurocognitive vulnerabilities, already from an early age onward. Neurobehavioral and neurocognitive difficulties tended to become more pronounced with increasing age and were rather robust, independent of specific karyotype, pre/postnatal diagnosis, or ascertainment strategy. A more longitudinal perspective on neurodevelopmental 'at-risk' pathways is warranted, also including studies assessing the effectiveness of targeted early interventions. Neurocognitive markers that signal differences in neurodevelopment may prove to be helpful in this. Focusing on early development of language, social cognition, emotion regulation, and executive functioning may help in uncovering early essential mechanisms of (later) neurobehavioral outcome, allowing for more targeted support and early intervention. This work is licensed under a Creative Commons Attribution-NonCommercial-NoDerivatives 4.0 International License.

Neurocognitive and behavioral development in young children (1–7 years) with sex chromosome trisomy

Endocrine Connections

Investigating sex chromosome trisomies (SCTs) may help in understanding neurodevelopmental pathwa... more Investigating sex chromosome trisomies (SCTs) may help in understanding neurodevelopmental pathways underlying the risk for neurobehavioral problems and psychopathology. Knowledge about the neurobehavioral phenotype is needed to improve clinical care and early intervention for children with SCT. This is especially relevant considering the increasing number of early diagnosed children with the recent introduction of noninvasive prenatal screening. The TRIXY Early Childhood Study is a longitudinal study designed to identify early neurodevelopmental risks in children with SCT, aged 1–7 years. This review summarizes the results from the TRIXY Early Childhood Study, focusing on early behavioral symptoms in areas of autism spectrum disorder, attention-deficit hyperactivity disorder, and communication disorders, and underlying neurocognitive mechanisms in domains of language, emotion regulation, executive functioning, and social cognition. Behavioral symptoms were assessed through structur...

The impact of sex chromosome trisomies (XXX, XXY, XYY) on gaze towards faces and affect recognition: a cross-sectional eye tracking study

Journal of Neurodevelopmental Disorders, Aug 2, 2022

The Impact of Sex Chromosome Trisomies (XXX, XXY, XYY) on Social Attention and Affect Recognition: A Cross-Sectional Eye Tracking Study

Background About 1:650–1000 children are born with an extra X or Y chromosome (47,XXX; 47,XXY; 47... more Background About 1:650–1000 children are born with an extra X or Y chromosome (47,XXX; 47,XXY; 47,XYY), which results in a Sex Chromosome Trisomy (SCT). This international cross-sectional study was designed to investigate social attention and affect recognition during early life of children with SCT, with the aim to find indicators for support and treatment. Methods A group of 101 children with SCT (aged 1–7 years old; Mage= 3.7 years) was included in this study, as well as a population-based sample of 98 children without SCT (Mage= 3.7). Social attention was measured using an eye tracking method that quantifies first fixations and fixation durations on eyes of static faces and fixation durations on social information (eyes, faces) in a dynamic paradigm (with two conditions: single face and multiple faces). Affect recognition was measured using the subtest Affect Recognition of the NEPSY-II neuropsychological test battery. Recruitment and assessment took place in the Netherlands and...

Early Social Behavior in Young Children with Sex Chromosome Trisomies (XXX, XXY, XYY): Profiles of Observed Social Interactions and Social Impairments Associated with Autism Spectrum Disorder (ASD)

Journal of Autism and Developmental Disorders

Individuals with Sex Chromosome Trisomies (SCT; XXX, XXY, XYY) have an increased vulnerability fo... more Individuals with Sex Chromosome Trisomies (SCT; XXX, XXY, XYY) have an increased vulnerability for developing challenges in social adaptive functioning. The present study investigates social interaction behavior in the context of varying social load, and Autism Spectrum Disorder (ASD) symptomatology in young children aged 1–7.5 years old, with SCT (N = 105) and control children (N = 101). Children with SCT show less interaction behaviors and more social withdrawal, as compared to their control peers, which were most evident in the high social load condition. Second, social impairments related to ASD are more prevalent, as compared to controls (27.1% at clinical level). These findings stress the importance of early monitoring and (preventive) support of early social development in young children with SCT.

Early impact of X‐ and Y‐chromosome variations ( XXX , XXY , XYY ) on social communication and social emotional development in 1–2‐year‐old children

American Journal of Medical Genetics Part A

Early Preventive Intervention for Young Children With Sex Chromosome Trisomies (XXX, XXY, XYY): Supporting Social Cognitive Development Using a Neurocognitive Training Program Targeting Facial Emotion Understanding

Frontiers in Psychiatry

BackgroundSex Chromosome Trisomies (SCTs; XXX, XXY, XYY) are genetic conditions that are associat... more 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 inc...

The Impact of Sex Chromosome Trisomies (XXX, XXY, XYY) on Social Attention and Affect Recognition: A Cross-Sectional Eye Tracking Study

Research Square (Research Square), Jun 21, 2021

Background About 1:650-1000 children are born with an extra X or Y chromosome (47,XXX; 47,XXY; 47... more Background About 1:650-1000 children are born with an extra X or Y chromosome (47,XXX; 47,XXY; 47,XYY), which results in a Sex Chromosome Trisomy (SCT). This international cross-sectional study was designed to investigate social attention and affect recognition during early life of children with SCT, with the aim to nd indicators for support and treatment. Methods A group of 101 children with SCT (aged 1-7 years old; M age = 3.7 years) was included in this study, as well as a population-based sample of 98 children without SCT (M age = 3.7). Social attention was measured using an eye tracking method that quanti es rst xations and xation durations on eyes of static faces and xation durations on social information (eyes, faces) in a dynamic paradigm (with two conditions: single face and multiple faces). Affect recognition was measured using the subtest Affect Recognition of the NEPSY-II neuropsychological test battery. Recruitment and assessment took place in the Netherlands and the United States. Results Eyetracking results reveal that children with SCT show lower proportions xation duration on social information already from the age of three years, compared to children without SCT. Also, impairments in the clinical range for affect recognition were found (32.2% of the SCT group scored in the well below average range); these di culties increase with age. Conclusions These results highlight the importance to further explore the development of social cognition skills of children with SCT in a longitudinal design, the monitoring of affect recognition skills, and the implementation of (preventive) interventions aiming to support the development of social attention and affect recognition.

Early impact of X‐ and Y‐chromosome variations ( <scp>XXX</scp> , <scp>XXY</scp> , <scp>XYY</scp> ) on social communication and social emotional development in 1–2‐year‐old children

American Journal of Medical Genetics, Mar 14, 2022

Sex chromosome trisomies (SCTs) are characterized by an extra X-or Y-chromosome (XXX, XXY, XYY). ... more Sex chromosome trisomies (SCTs) are characterized by an extra X-or Y-chromosome (XXX, XXY, XYY). The present study aims to investigate early signs of social communication and social emotional development in very young children with SCT. Thirty-four children with SCT (aged 12-24 months) were included in this study, as well as 31 agematched controls. Social communication was measured with structured behavior observations according to the Early Social Communication Scales, and social emotional developmental level with the Bayley Social Emotional parental questionnaire. Recruitment and assessment took place in the Netherlands and in the United States. On average, 12-24-month old children with SCT showed difficulties with early social communication, more so in responding to others as compared to initiating social communications. During social interactions, children with SCT made less frequent eye contact, compared to controls. Also, difficulties in acquiring social emotional milestones were found in 1-year old children with SCT, with 44% of the children having social emotional vulnerabilities in the borderline or extremely low range, compared to typically developing children. In this cohort, no significant predictive effects of karyotype-subtype (XXX, XXY, XYY) were found. Already from a very early age, SCT can be associated with increased risk for vulnerabilities in adaptive social functioning. These findings suggest that SCT impact the maturation of the social brain already from an early age, and stress the importance of early monitoring and (preventive) support early social development in young children with SCT.

The impact of sex chromosome trisomies (XXX, XXY, XYY) on gaze towards faces and affect recognition: a cross-sectional eye tracking study

Journal of Neurodevelopmental Disorders, Aug 2, 2022

Background: About 1:650-1000 children are born with an extra X or Y chromosome (47,XXX; 47,XXY; 4... more Background: About 1:650-1000 children are born with an extra X or Y chromosome (47,XXX; 47,XXY; 47,XYY), which results in a sex chromosome trisomy (SCT). This international cross-sectional study was designed to investigate gaze towards faces and affect recognition during early life of children with SCT, with the aim to find indicators for support and treatment. Methods: A group of 101 children with SCT (aged 1-7 years old; M age = 3.7 years) was included in this study, as well as a population-based sample of 98 children without SCT (M age = 3.7). Eye gaze patterns to faces were measured using an eye tracking method that quantifies first fixations and fixation durations on eyes of static faces and fixation durations on eyes and faces in a dynamic paradigm (with two conditions: single face and multiple faces). Affect recognition was measured using the subtest Affect Recognition of the NEPSY-II neuropsychological test battery. Recruitment and assessment took place in the Netherlands and the USA. Results: Eye tracking results reveal that children with SCT show lower proportion fixation duration on faces already from the age of 3 years, compared to children without SCT. Also, impairments in the clinical range for affect recognition were found (32.2% of the SCT group scored in the well below average range). Conclusions: These results highlight the importance to further explore the development of social cognitive skills of children with SCT in a longitudinal design, the monitoring of affect recognition skills, and the implementation of (preventive) interventions aiming to support the development of attention to social important information and affect recognition.

Early Social Behavior in Young Children with Sex Chromosome Trisomies (XXX, XXY, XYY): Profiles of Observed Social Interactions and Social Impairments Associated with Autism Spectrum Disorder (ASD)

Journal of Autism and Developmental Disorders, May 12, 2022

Individuals with Sex Chromosome Trisomies (SCT; XXX, XXY, XYY) have an increased vulnerability fo... more Individuals with Sex Chromosome Trisomies (SCT; XXX, XXY, XYY) have an increased vulnerability for developing challenges in social adaptive functioning. The present study investigates social interaction behavior in the context of varying social load, and Autism Spectrum Disorder (ASD) symptomatology in young children aged 1-7.5 years old, with SCT (N = 105) and control children (N = 101). Children with SCT show less interaction behaviors and more social withdrawal, as compared to their control peers, which were most evident in the high social load condition. Second, social impairments related to ASD are more prevalent, as compared to controls (27.1% at clinical level). These findings stress the importance of early monitoring and (preventive) support of early social development in young children with SCT.

Early symptoms of autism spectrum disorder (ASD) in 1–8 year old children with sex chromosome trisomies (XXX, XXY, XYY), and the predictive value of joint attention

European Child & Adolescent Psychiatry, Sep 15, 2022

The objective of the present study is to investigate the impact of Sex Chromosome Trisomy (SCT; X... more The objective of the present study is to investigate the impact of Sex Chromosome Trisomy (SCT; XXX, XXY, XYY) on the early appearance of Autism Spectrum Disorder (ASD) symptoms, and the predictive value of Joint Attention for symptoms of ASD. SCTs are specific genetic conditions that may serve as naturalistic 'at risk' models of neurodevelopment, as they are associated with increased risk for neurobehavioral vulnerabilities. A group of 82 children with SCT (aged 1-8 years) was included at baseline of this longitudinal study. Joint Attention was measured at baseline with structured behavior observations according to the Early Social Communication Scales. ASD symptoms were assessed with the Modified Checklist for Autism in Toddlers questionnaire and Autism Diagnostic Interview-Revised in a 1-year follow-up. Recruitment and assessment took place in the Netherlands and in the United States. The results demonstrate that ASD symptoms were substantially higher in children with SCT compared to the general population, with 22% of our cohort at clinical risk for ASD, especially in the domain of social interaction and communication. Second, a predictive value of Joint Attention was found for ASD symptoms at 1-year follow-up. In this cohort, no differences were found between karyotype-subtypes. In conclusion, from a very early age, SCT can be associated with an increased risk for vulnerabilities in adaptive social functioning. These findings show a neurodevelopmental impact of the extra X or Y chromosome on social adaptive development associated with risk for ASD already from early childhood onward. These findings advocate for close monitoring and early (preventive) support, aimed to optimize social development of young children with SCT.

The Impact of Sex Chromosome Trisomies (XXX, XXY, XYY) on Early Social Cognition: Social Orienting, Joint Attention, and Theory of Mind

Archives of Clinical Neuropsychology, 2021

Objective About 1:650–1,000 children are born with an extra X or Y chromosome (XXX; XXY; XYY), wh... more Objective About 1:650–1,000 children are born with an extra X or Y chromosome (XXX; XXY; XYY), which results in a sex chromosome trisomy (SCT). This study aims to cross-sectionally investigate the impact of SCT on early social cognitive skills. Basic orienting toward social cues, joint attention, and theory of mind (ToM) in young children with SCT were evaluated. Method About 105 children with SCT (range: 1–7 years old) were included in this study, as well as 96 age-matched nonclinical controls. Eyetracking paradigms were used to investigate the eye gaze patterns indicative of joint attention skills and orienting to social interactions. The ToM abilities were measured using the subtest ToM of the Developmental NEuroPSYchological Assessment, second edition, neuropsychological test battery. Recruitment and assessment took place in the Netherlands and in the United States. Results Eyetracking results revealed difficulties in children with SCT in social orienting. These difficulties wer...

Mindfulness-Based Stress Reduction in Prison: Experiences of Inmates, Instructors, and Prison Staff

International Journal of Offender Therapy and Comparative Criminology, 2019

Mindfulness intervention aims to reduce stress and to improve physical and mental health. The pre... more Mindfulness intervention aims to reduce stress and to improve physical and mental health. The present study investigated feasibility and effectiveness of mindfulness intervention in a prison context, in both a qualitative and quantitative fashion. Specifically, the mindfulness-based stress reduction (MBSR) intervention was investigated, in a retrospective pre–post design, in five Dutch prisons. Twenty-two inmates (out of 25 approached, mean age: 40.1 years ( SD = 11.1), convicted of murder, manslaughter, sexual offenses, drug offenses, robbery with violence, and/or illegal restraint/kidnap, and sentenced to incarceration between 15 and 209 months ( M = 5.5 years; SD = 3.8) took part in a semistructured interview after completion of the MBSR intervention. The interviews addressed level of satisfaction and challenges regarding the MBSR intervention as well as potential effects on stress responsivity, coping style, impulse control, aggression, and self-esteem. Ten staff members and fou...

Empathic distress and concern predict aggression in toddlerhood: The moderating role of sex

Infant Behavior and Development, 2019

This is an open access article under the terms of the Creat ive Commo ns Attri bution-NonCo mmerc... more

Neurocognitive and behavioral development in young children (1-7 years) with sex chromosome trisomy

Endocrine connections, Mar 1, 2023

Investigating sex chromosome trisomies (SCTs) may help in understanding neurodevelopmental pathwa... more Investigating sex chromosome trisomies (SCTs) may help in understanding neurodevelopmental pathways underlying the risk for neurobehavioral problems and psychopathology. Knowledge about the neurobehavioral phenotype is needed to improve clinical care and early intervention for children with SCT. This is especially relevant considering the increasing number of early diagnosed children with the recent introduction of noninvasive prenatal screening. The TRIXY Early Childhood Study is a longitudinal study designed to identify early neurodevelopmental risks in children with SCT, aged 1-7 years. This review summarizes the results from the TRIXY Early Childhood Study, focusing on early behavioral symptoms in areas of autism spectrum disorder, attention-deficit hyperactivity disorder, and communication disorders, and underlying neurocognitive mechanisms in domains of language, emotion regulation, executive functioning, and social cognition. Behavioral symptoms were assessed through structured behavior observation and parental questionnaires. Neurocognition was measured using performance tests, eyetracking, and psychophysiological measures of arousal. In total, 209 children aged 1-7 years were included: 107 children with SCT (33 XXX, 50 XXY, and 24 XYY) and 102 age-matched population controls. Study outcomes showed early behavioral symptoms in young children with SCT, and neurocognitive vulnerabilities, already from an early age onward. Neurobehavioral and neurocognitive difficulties tended to become more pronounced with increasing age and were rather robust, independent of specific karyotype, pre/postnatal diagnosis, or ascertainment strategy. A more longitudinal perspective on neurodevelopmental 'at-risk' pathways is warranted, also including studies assessing the effectiveness of targeted early interventions. Neurocognitive markers that signal differences in neurodevelopment may prove to be helpful in this. Focusing on early development of language, social cognition, emotion regulation, and executive functioning may help in uncovering early essential mechanisms of (later) neurobehavioral outcome, allowing for more targeted support and early intervention. This work is licensed under a Creative Commons Attribution-NonCommercial-NoDerivatives 4.0 International License.

Neurocognitive and behavioral development in young children (1–7 years) with sex chromosome trisomy

Endocrine Connections

Investigating sex chromosome trisomies (SCTs) may help in understanding neurodevelopmental pathwa... more Investigating sex chromosome trisomies (SCTs) may help in understanding neurodevelopmental pathways underlying the risk for neurobehavioral problems and psychopathology. Knowledge about the neurobehavioral phenotype is needed to improve clinical care and early intervention for children with SCT. This is especially relevant considering the increasing number of early diagnosed children with the recent introduction of noninvasive prenatal screening. The TRIXY Early Childhood Study is a longitudinal study designed to identify early neurodevelopmental risks in children with SCT, aged 1–7 years. This review summarizes the results from the TRIXY Early Childhood Study, focusing on early behavioral symptoms in areas of autism spectrum disorder, attention-deficit hyperactivity disorder, and communication disorders, and underlying neurocognitive mechanisms in domains of language, emotion regulation, executive functioning, and social cognition. Behavioral symptoms were assessed through structur...

The impact of sex chromosome trisomies (XXX, XXY, XYY) on gaze towards faces and affect recognition: a cross-sectional eye tracking study

Journal of Neurodevelopmental Disorders, Aug 2, 2022

The Impact of Sex Chromosome Trisomies (XXX, XXY, XYY) on Social Attention and Affect Recognition: A Cross-Sectional Eye Tracking Study

Background About 1:650–1000 children are born with an extra X or Y chromosome (47,XXX; 47,XXY; 47... more Background About 1:650–1000 children are born with an extra X or Y chromosome (47,XXX; 47,XXY; 47,XYY), which results in a Sex Chromosome Trisomy (SCT). This international cross-sectional study was designed to investigate social attention and affect recognition during early life of children with SCT, with the aim to find indicators for support and treatment. Methods A group of 101 children with SCT (aged 1–7 years old; Mage= 3.7 years) was included in this study, as well as a population-based sample of 98 children without SCT (Mage= 3.7). Social attention was measured using an eye tracking method that quantifies first fixations and fixation durations on eyes of static faces and fixation durations on social information (eyes, faces) in a dynamic paradigm (with two conditions: single face and multiple faces). Affect recognition was measured using the subtest Affect Recognition of the NEPSY-II neuropsychological test battery. Recruitment and assessment took place in the Netherlands and...

Early Social Behavior in Young Children with Sex Chromosome Trisomies (XXX, XXY, XYY): Profiles of Observed Social Interactions and Social Impairments Associated with Autism Spectrum Disorder (ASD)

Journal of Autism and Developmental Disorders

Individuals with Sex Chromosome Trisomies (SCT; XXX, XXY, XYY) have an increased vulnerability fo... more Individuals with Sex Chromosome Trisomies (SCT; XXX, XXY, XYY) have an increased vulnerability for developing challenges in social adaptive functioning. The present study investigates social interaction behavior in the context of varying social load, and Autism Spectrum Disorder (ASD) symptomatology in young children aged 1–7.5 years old, with SCT (N = 105) and control children (N = 101). Children with SCT show less interaction behaviors and more social withdrawal, as compared to their control peers, which were most evident in the high social load condition. Second, social impairments related to ASD are more prevalent, as compared to controls (27.1% at clinical level). These findings stress the importance of early monitoring and (preventive) support of early social development in young children with SCT.

Early impact of X‐ and Y‐chromosome variations ( XXX , XXY , XYY ) on social communication and social emotional development in 1–2‐year‐old children

American Journal of Medical Genetics Part A

Early Preventive Intervention for Young Children With Sex Chromosome Trisomies (XXX, XXY, XYY): Supporting Social Cognitive Development Using a Neurocognitive Training Program Targeting Facial Emotion Understanding

Frontiers in Psychiatry

BackgroundSex Chromosome Trisomies (SCTs; XXX, XXY, XYY) are genetic conditions that are associat... more 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 inc...