Association Between Challenging Behaviour and Sleep Problems in Adults Enrolled in the Global Angelman Syndrome Registry.

Angelman Syndrome (AS) is a rare genetic disorder that impacts 1:20,000 people. Challenging behaviour, such as severe injurious behaviour, aggression and frequent unprovoked episodes of laughter are a significant problem among adults with AS that adversely impacts an individual's quality of life. This study, for the first time, aims understand the characteristic of challenging behaviour, its frequency, and the factors associated with it in adults with AS. Data from participants with AS (N = 37; aged 18-46 years) registered with the Global Angelman Registry, were divided into challenging behaviour and non-challenging behaviour groups based on the presence or absence of 50% of the behaviours recorded in the registry. Descriptive statistics, chi-squared and t-test analysis were conducted to assess the impact of variables on challenging behaviour. Multiple regressions were conducted to investigate the predictors of challenging behaviour. 56% of the sample presented with challenging behaviour. Disorders of arousal, self-injury, behaviour dysregulation, repetitive behaviour, and the lack of physical therapy accounted for 59% of the variance of challenging behaviour in this population. It was found that challenging behaviour was very common in this population. A significant association was found between challenging behaviour and both sleep arousal and the lack of physical therapy. Sleep arousal and the lack of physical therapy were the key factors associated with challenging behaviour in this study. Targeted interventions are needed to decrease challenging behaviour and future research should focus on sleep interventions and increased opportunities for physical therapy.

Factors associated with sleep disturbances in children and adolescents with Angelman Syndrome.

BACKGROUND: Angelman Syndrome (AS) is a rare genetic disorder characterised by hyperactivity, overexcitability, developmental delays, and lack of speech. METHODS: This study used secondary data analysis to investigate sleep disturbances in children and adolescents (n = 212) who are enrolled in the Global Angelman Syndrome Registry. Participants were divided into two groups based on the presence or absence of sleep disturbance. The cut-off score of 40 on the Sleep Disturbance Scale for Children was used to indicate the presence or absence of sleep disturbances. Sleep disturbances and their association with co-occurring conditions were examined regarding challenging behaviour, language and communication, infancy history, gastrointestinal symptoms, and epilepsy. Multiple regression was then conducted to investigate possible predictors for sleep disturbances. RESULTS: Children and adolescents with AS, with and without sleep disturbances, differed considerably regarding anxiety. Sleep disturbances were significantly associated with an ability to use spoken words and computerised communication devices, and anxiety was a predictor of sleep disturbances. CONCLUSION: Future research is necessary to replicate this novel research, and to advance the clinical treatment of sleep disturbances in children and adolescents with AS.

Relationship Between Gastrointestinal Symptoms in Autism Spectrum Disorder and Parent Stress, Anxiety, Depression, Quality of Life and Social Support.

Gastrointestinal (GI) symptoms are a common comorbidity in children and adolescents with autism spectrum disorder (ASD). Little is known about the impact that GI symptoms have on parental well-being. Parents of 409 children and adolescents with ASD completed the GI Symptoms Inventory, Parenting Stress Index-Short Form, World Health Organization Quality of Life Abbreviated Version, Hospital Anxiety and Depression Scale, and the Multidimensional Scale of Perceived Social Support. High levels of stress were demonstrated by parents with 40.1% receiving clinically significant scores. A relationship was found between parental stress and GI symptoms. Parental anxiety and depression were found at high levels but were not more common in parents of individuals with GI symptoms than those without. Lower levels of quality of life were found in parents of individuals with GI symptoms compared to parents of individuals without GI symptoms. Parents of children with GI symptoms were less satisfied with their personal and social relationships with others. Parents of children with GI symptoms had lower scores on a measure of perceived social support than parents of children and adolescents without GI symptoms. GI symptoms are stressful for parents and future research is needed to determine how to alleviate this stress and to improve the quality of life of parents of individuals with ASD.

Relationship between child sleep problems in autism spectrum disorder and parent mental health and well-being.

Sleep problems are a common comorbidity in children and adolescents with autism spectrum disorder (ASD). The aim of this study is to determine how sleep problems affect not only the child with ASD, but parents also. Parents of 409 children and adolescents with ASD completed the Children's Sleep Habits Questionnaire, Pittsburgh Sleep Quality Index, Parenting Stress Index-Short Form, World Health Organization Quality of Life Abbreviated Version (WHOQOL-BREF), Hospital Anxiety and Depression Scale, and the Multidimensional Scale of Perceived Social Support. The majority (86.6%) of parents presented with poor sleep. The majority (95.3%; n = 387) of children presented with sleep problems, while 4.7% (n = 22) did not have sleep problems. A cross-sectional within-subjects research design was utilised, with the following analyses conducted: Pearson correlations, chi-square tests, t-tests, and MANOVAs. Relationships were found between child sleep problems and parent sleep problems, specifically child parasomnias, sleep duration, night wakings, and sleep onset delay. Parents of children with sleep problems experienced more parenting stress, specifically on the Difficult Child and Parent-Child Dysfunctional Interaction subscales of the Parenting Stress Index-Short Form. Parents of children and adolescents with sleep problems had significantly higher levels of anxiety and depression than parents of children and adolescents who did not have sleep problems. A relationship between sleep problems and lower quality of life was found. Parents of children with sleep problems received significantly lower scores on the WHOQOL-BREF domains of Physical Health, Psychological and Environment than parents of children without sleep problems. There was no significant difference found between parents of children with or without sleep problems on perceived social support. The current study demonstrated how child sleep affects parental well-being. While sleep problems are one comorbid condition in ASD, future research is needed to determine the impact of other comorbidities in parents of children and adolescents with ASD.

A delayed intervention start randomized controlled trial of high- and low-tech communication training approaches for school-age autistic children with co-occurring intellectual disability.

The researchers designed a delayed intervention start randomized controlled trial to compare improvements in functional communication following augmentative and alternative communication (AAC) interventions. The study compared outcomes from function-based applied behavior analytic (ABA) and eclectic non-ABA forms of classroom-based communication strategies (waitlist control) as well as from high- and low-tech forms of AAC. High-tech AAC consisted of tablet-based communication, and low-tech AAC used an exchange of picture cards. The community-based sample consisted of 29 autistic children with a co-occurring intellectual disability. Participants were randomized to groups (AAC, waitlist control), and each group received approximately 3 months of communication intervention. Multilevel modeling of learner outcomes indicated that the function-based approach produced greater improvements than the eclectic alternative, but significant differences were not observed between outcomes of high- and low-tech forms of function-based AAC. These results are consistent with earlier investigations and provide supporting evidence that both high- and low-tech forms of function-based intervention are effective for use with autistic children with accompanying intellectual disability. Additional discussion is provided regarding further research into how technology is applied and incorporated into behavior analytic programming.

Adaptive living skills, sleep problems, and mental health disorders in adults with 22q11.21 deletion syndrome.

BACKGROUND: 22q11.21 deletion syndrome (22q11DS) is a neurodevelopmental syndrome caused by a microdeletion of genes at the 22q11.21 locus. It has a prevalence of 1:2000. This study investigated the prevalence of adaptive living skills, sleep problems, and mental health disorders in adults with 22q11DS and examined the relationship between these factors. METHODS: Parents with an adult son or daughter with 22q11DS completed the following: A bespoke Demographic Information Questionnaire, Sleep Questionnaire (SQ-SP), Psychopathology in Autism Checklist (PAC), and Activities of Daily Living (ADL) scale. Descriptive statistics, correlations, and one-way between groups analysis of variance (ANOVA) were conducted. RESULTS: Mental health difficulties, sleep problems, and low levels of adaptive living skills are prevalent in adults with 22q11DS. Strong positive correlations were identified between sleep problems, depression, and anxiety subscale scores and moderate negative correlations between depression, psychosis, and activities of daily living skills. CONCLUSION: Adults with 22q11DS need screening and treatment for mental health and sleep problems.

Adaptation of Parents Raising a Child with ASD: The Role of Positive Perceptions, Coping, Self-efficacy, and Social Support.

This study explored the adaptation of parents raising a child with an Autism Spectrum Disorder (ASD) specifically the contributory role of positive perceptions, coping, self-efficacy, and social support. One hundred and thirty-six parents of children with a diagnosis of ASD completed a battery of self-report questionnaires via an online survey. Using multiple regression analyses positive perceptions, adaptive coping, self-efficacy, and social support were each a significant contributor to one or more positive adaptation outcomes. Multiple moderated regression analysis found no evidence that these factors were significant moderators between behavioural problems and parental adaptation. The implications of these findings in supporting parents raising a child with ASD are outlined.

Relationships between challenging behavior and gastrointestinal symptoms, sleep problems, and internalizing and externalizing symptoms in children and adolescents with Angelman syndrome.

BACKGROUND: Angelman syndrome (AS), is a rare genetic disorder. This study investigated the relationship between parent-reported comorbid symptoms including gastrointestinal symptoms, sleep problems, internalizing symptoms, and behavior problems in children and adolescents with AS. METHOD: Parents of 98 children and adolescents with AS completed the Gastrointestinal Symptom Inventory, Children's Sleep Habits Questionnaire, Child Behavior Checklist, Social Communication Questionnaire, and the Behavior Problem Inventory-Short Form. Data were analyzed using descriptive statistics, Pearson's correlation coefficients, and hierarchical multiple regressions. RESULTS: There was a high frequency of GI symptoms (99%), sleep problems (95.9%), challenging behavior (98%), internalizing symptoms (38%), and 72.4% of children and adolescents presented with ASD symptoms. Self-injurious behavior (SIB), aggressive/destructive behavior, and the frequency of stereotyped behavior positively correlated with GI symptoms and sleep problems and it was moderately negatively associated with age. Internalizing symptoms and age were positively associated with SIB. Aggression was significantly related to gender, but not the presence of ASD symptoms. CONCLUSIONS: Findings highlight the relationships between comorbid conditions. They may lead to a deeper understanding of how comorbidities present in children and adolescents with Angelman Syndrome.

Examining the Relationship Between Sleep Quality, Social Functioning, and Behavior Problems in Children with Autism Spectrum Disorder: A Systematic Review.

Over forty percent of autistic children experience poor sleep quality, and social interaction difficulties are a core characteristic of autism. However, the relationship between sleep quality and social functioning and behavior remains poorly understood. This systematic review examined the evidence concerning the impact of sleep quality on the social functioning and behavior problems in autistic children and adolescents. It also identified key related factors and evaluated how this issue has been researched to date. Seven key journals were hand-searched and five databases were systematically searched, using keywords. Titles and abstracts of 4123 items were screened against eligibility criteria by two researchers. Relevant studies were retained if they were peer-reviewed empirical papers, published in English between 2000 and 2021. Then, the full text of 97 papers was screened and if they met the eligibility criteria, their reference lists were hand-searched. Forty-six studies were included in the final review. Data were systematically extracted and two authors critically appraised the strengths and weaknesses of studies using Critical Appraisal Skills Programme tools. Key themes were identified, because a meta-analysis was not possible due to the studies' heterogeneity. The review identified that sleep quality and social functioning are associated with one another and there is a small amount of evidence that a bi-directional causal relationship may exist. Evidence suggests that several nights of suboptimal sleep duration and a lack of deep continuous sleep negatively impact externalizing and internalizing behavior. Sleep quality is also reduced by anxiety and sensory sensitivity. However, longitudinal studies with larger samples are needed to establish causality. Future research needs to examine confounding factors and to develop consensus regarding best-practice processes for the objective measurement of sleep with autistic children. Additional research also needs to further examine the consequences of poor sleep quality on internalizing behavior, and the impact of socio-cultural practices.

Gastrointestinal Symptoms in Autism Spectrum Disorder: A Systematic Review.

This systematic review aims to offer an updated understanding of the relationship between gastrointestinal symptoms (GIS) and autism spectrum disorder (ASD) in children and adolescents. The databases PsycINFO, Medline, Cinahl, and ERIC were searched using keywords, and relevant literature was hand-searched. Papers (n = 3319) were systematically screened and deemed eligible if they were empirical studies published in English since 2014 and measured the GIS of individuals with ASD who were under 18 years old. Thirty studies were included in the final review. The study findings were synthesized under eight themes, including the prevalence and nature of GIS and their relationship with developmental regression, language and communication, ASD severity, challenging behavior, comorbid psychopathology, sleep problems, and sensory issues. The review found that GIS were common and that there was contradictory evidence concerning their relationship with co-occurring conditions. It also identified evidence of some causal relationships that support the existence of the gut-immune-brain pathways. Future research needs to use large prospective designs and objective and standardized GIS measurements to provide a nuanced understanding of GIS in the context of ASD.

Relationship between Comorbid Psychopathology in Children and Adolescents with Autism Spectrum Disorder and Parental Well-being.

AIM: Frequency and relationship between child comorbid psychopathology and parental stress, quality of life (QoL), anxiety, depression, and social support were examined in parents of children and adolescents with autism spectrum disorder (ASD). METHOD: Parents of 152 children and adolescents with ASD completed the Autism Spectrum Disorder-Comorbid for Children, Parenting Stress Index-Short Form, World Health Organization Quality of Life Abbreviated Version, Hospital Anxiety and Depression Scale, and the Multidimensional Scale of Perceived Social Support. RESULTS: A series of one-way multivariate analysis of variance was conducted to examine the relationship between child comorbid psychopathology and parental well-being. A relationship was found between parental QoL, depression and anxiety and child comorbid psychopathology. Results showed a relationship between parental stress and the severity of child conduct, and repetitive behaviors. CONCLUSION: This study adds to existing literature by demonstrating the relationship between comorbid psychopathology in children and adolescents in ASD and parental well-being.

Attention deficit hyperactivity disorder (ADHD) symptoms, comorbid psychopathology, behaviour problems and gastrointestinal symptoms in children and adolescents with autism spectrum disorder.

OBJECTIVES: The study aims to investigate attention deficit hyperactivity disorder (ADHD) symptoms, gastrointestinal (GI) symptoms, comorbid psychopathology and behaviour problems in children and adolescents with autism spectrum disorder (ASD). METHODS: Parents of 147 children and adolescents with ASD aged 6-18 years completed the Conners 3 Parent-Short Form, Gastrointestinal Symptom Inventory, Behavior Problems Inventory-Short Form and Autism Spectrum Disorder-Comorbid for Children. RESULTS: Fifty-six per cent of children and adolescents had a comorbid diagnosis of ADHD, yet over 70% presented with clinically significant ADHD symptoms. Forty per cent of participants received a diagnosis of ADHD before ASD and 25.6% received a diagnosis of ASD first. Relationships were found between ADHD symptoms and comorbid psychopathology, GI symptoms, and behaviour problems. CONCLUSIONS: The outcomes suggest that ADHD is being underestimated as a comorbid disorder of ASD. This may have implications on treatment and interventions for children and adolescents who have a diagnosis of both ASD and ADHD.

Familial sleep and autism spectrum disorder: a pilot actigraphy study of sleep quality, quality of life and psychological distress.

OBJECTIVES: Sleep problems are common among children with autism spectrum disorder (ASD) and can have a negative impact on the child's behaviour and daytime functioning. The current pilot study examined objective measurements of child and parent sleep as factors associated with the stress, anxiety, depressive symptoms, social support and quality of life of parents of children with ASD. METHODS: Participants were nine children with ASD and their parents (nine mothers and three fathers). Participants wore an actigraph for 7 consecutive days and nights. Measures of sleep habits and quality were used to ascertain child and parent sleep. Measures of parenting stress, anxiety, depressive symptoms, quality of life and social support were collated. RESULTS: Results indicated the emergence of high parental stress, anxiety and depressive symptoms. Significant correlations were observed between parent depressive symptoms, and both subjective sleep quality and child sleep disruptions. CONCLUSIONS: The present study found that parental well-being is affected by child sleep problems.

Age of Autism Spectrum Disorder Diagnosis and Comorbidity in Children and Adolescents with Autism Spectrum Disorder.

AIM: Research is required to study the relationship between age of autism spectrum disorder (ASD) diagnosis and the presence of comorbidities. METHOD: The Gastrointestinal Symptom Inventory, Autism Spectrum Disorder-Comorbid for Children, Behavior Problem Inventory-Short Form and Social Communication Questionnaire were completed by parents of 129 children and adolescents with a diagnosis of ASD. RESULTS: Results revealed significant relationships between the age of ASD diagnosis, the presence of comorbidities and intellectual disability. Significant correlations were found between the age of ASD diagnosis and self-injurious and stereotyped behavior. Comorbid psychopathology significantly predicted the presence of GI symptoms. In addition, the relationship between comorbid psychopathology and challenging behavior in this study was reported as bi-directional as both comorbidities predicted one another in the sample. CONCLUSION: Future research needs to consider the role of comorbidities in relation to ASD diagnosis.

Attention-Deficit/Hyperactivity Disorder Symptoms, Gastrointestinal Symptoms, Sleep Problems, Challenging Behavior, Adaptive Behavior, and Quality of Life in Children and Adolescents with Autism Spectrum Disorder.

This study investigated the relationship between sleep, gastrointestinal symptoms, challenging behavior, adaptive behavior, and quality of life between children and adolescents with autism spectrum disorder (ASD), with and without attention-deficit/hyperactivity disorder (AD/HD) symptoms. Parents of 118 children and adolescents with ASD completed the Conners Early Childhood Rating Scale-Parent Short Form or the Conners 3-Parent Short Form, Children's Sleep Habits Questionnaire, Gastrointestinal Symptom Inventory, Behavior Problems Inventory-Short Form, Pediatric Quality of Life Inventory and the Vineland Adaptive Behavior Scales, Second Edition. The ASD group and the ASD with AD/HD groups differed significantly in sleep problems, gastrointestinal symptoms, and quality of life. Regressions indicated that AD/HD symptoms accounted for a small proportion of the variance for the differences in sleep problems and quality of life. AD/HD symptoms contribute to the complex needs of individuals with ASD. Research is necessary to investigate how these symptoms exacerbate comorbidities.

Gaming Disorder in Adults with Autism Spectrum Disorder.

Gaming disorder (GD) is a clinical addiction to video or internet games. This study investigated whether GD symptoms are heightened in adults with autism spectrum disorder (ASD) in comparison to a control group, and explored predictors of GD in 230 adults with ASD and 272 controls. The relationship between GD and gelotophobia was examined. Measures included the Ten-Item Internet Gaming Disorder Test, GELOPH < 15 >, Autism Spectrum Quotient-10 items, Inventory of Parent and Peer attachment, Emotional Regulation Questionnaire, Social Functioning Questionnaire (SFQ) and the NEO-FFI-3. Individuals in the ASD group showed significantly higher symptoms of GD. Peer-attachment, emotional regulation and extraversion significantly predicted GD scores. Gelotophobia and GD were related to each other with a small effect size.

Transition Program from School to Employment in Youths with Intellectual Disability: Evaluation of the Irish Pilot Study E-IDEAS.

AIM: The purpose of this pilot was to evaluate the effectiveness of the E-IDEAS curriculum, specifically designed for workplace inclusion of youths with intellectual disabilities (ID) and aimed to transfer social, communication, independent living and employment skills. METHOD: The curriculum was attended by 5 participants, and it was provided concurrently with five different work-placements across a period of six months. Two assessment tools were used to demonstrate the acquisition of such skills and an evidence-base improvement of their quality of life. Pre and post-intervention standardized assessments were also taken for measuring the improvement in quality of life (through the San Martín Scale) and adaptive behavior (through the Vineland-II Adaptive Behavior Scale). RESULTS: The data collected showed increases in the acquisition of such skills. Evidence of maintenance and generalization were also demonstrated. CONCLUSION: Implications for practice and further research are discussed.

The Co-Occurrence of Autism Spectrum Disorder and Cerebral Palsy and Associated Comorbid Conditions in Children and Adolescents.

BACKGROUND: Comorbidity is the co-occurrence of two or more disorders in the same person. AIM: This study investigated the frequency of comorbid conditions, in children and adolescents, with autism spectrum disorder (ASD), cerebral palsy (CP), and a comorbid diagnosis of ASD and CP. METHOD: Ninety-six children and adolescents with ASD, CP, and both ASD and CP aged between 4 and 18 years participated in this study. Parents completed the Gastrointestinal Symptom Inventory, Children's Sleep Habits Questionnaire, Child Behavior Checklist, Social Communication Questionnaire, and the Vineland Adaptive Behavior Scales. RESULTS: Results of ANOVA analyses revealed significant group differences in sleep problems, social communication difficulties, and adaptive behavior. Regression analysis found that the presence of an intellectual disability significantly predicted levels of adaptive behavior. CONCLUSION: This research demonstrated the importance of studying comorbidities in children and adolescents with CP alone, ASD alone, and combined ASD and CP.

An examination of adaptive behavior and functional outcomes in adults with 22q11.2 deletion syndrome: A parental perspective.

22q11.2 Deletion Syndrome (22q11DS) is a genetic syndrome caused by a chromosomal microdeletion. It affects approximately 1 in 850-992 pregnancies, and its clinical manifestations include congenital heart disease, gastrointestinal symptoms, and psychiatric illnesses. The study examined the relationship between adaptive behavior and functional outcomes, educational attainment, employment, and independent living, and whether age, gender, intellectual disability, presence of psychiatric disorder, and close friendships could predict levels of adaptive behavior. Parents of adults with 22q11DS (n = 101; 48 male and 54 female) completed the Waisman Activities of Daily Living Scale, demographic details, and questions elicited employment, education, and relationships status. Analysis conducted in SPSS, included descriptive statistics, measures of association, Analysis of Variance, logistic and linear regressions. Differences in levels of overall adaptive behavior were found regarding employment and living status, but not in educational attainment. Having close friendships was associated with adaptive behavior as well as the likelihood of living independently. Further research is needed, ideally using prospective designs and purposive sampling strategies. This needs to examine how social and communication deficits impact relationship building and how they are affected by the clinical manifestations of 22q11DS. It also needs to focus on how different social structures interface with levels of adaptive behavior.

Comorbid Psychopathology, Challenging Behavior, Sensory Issues, Adaptive Behavior and Quality of Life in Children and Adolescents with Autism Spectrum Disorder.

Aim: Comorbid psychopathology refers to having a diagnosis of two or more co-occurring psychological disorders. The current study investigated the differences between children and adolescents with no-mild, moderate and severe comorbid psychopathology in children and adolescents with ASD.Method: Parents of 133 children completed the Autism Spectrum Disorder-Comorbid for Children, Behavior Problems Inventory-Short Form, Pediatric Quality of Life Inventory, Vineland Adaptive Behavior Scales, Social Communication Questionnaire, Short Sensory Profile, and Behavioral/Educational Interventions and Complementary/Alternative Medicine (CAM) Interventions of the Autism Treatment Network Registry Parent Baseline Assessment.Results: A significant difference was found between severity of comorbid psychopathology and all types of challenging behavior and all sensory issues except movement. A small effect size was also found between comorbid psychopathology and quality of life.Conclusion: The findings from this study show significant difficulties associated with those with comorbid psychopathology in ASD in challenging behavior, sensory issues and quality of life.