Caregiver-based perception of disease burden in Schaaf-Yang syndrome.

BACKGROUND: Schaaf-Yang syndrome (SYS) is a neurodevelopmental disorder caused by truncating variants in the paternally expressed MAGEL2 gene in the Prader-Willi syndrome-region on chromosome 15q. In addition to hypotonia and intellectual disability, individuals with SYS are frequently affected by neonatal contractures and autism spectrum disorder. In this study, we focus on the burden of disease on patients and their families for the first time. METHODS: Based on the online SYS Patient Voices Survey the perspective of 81 primary caregivers on SYS was assessed. RESULTS: The perceived severity of muscular and developmental manifestations dominated the evaluation of the phenotype in early childhood, while behavioral issues were considered more impactful later in life. Importantly, an apprehension toward symptoms with a later onset was observed in caregivers of younger children. Available therapeutic options, while mostly effective, did not sufficiently alleviate the total burden of disease. Overall, parents stated that caring for an individual with SYS was very challenging, affecting their daily lives and long-term planning. CONCLUSION: Our study demonstrates the necessity for treatments that, adapted to age and in accordance with the caregivers' prioritization, improve the patients' medical condition and thus facilitate their and their families' social participation.

Brief Report: Health Expenditures for Children with Autism and Family Financial Well-Being in China.

Little is known on the financial well-being of families raising children with autism spectrum disorders (ASD). Family financial well-being has important impacts on the development of children with ASD. The study uses a 2019 survey collected from Chinese families raising a child with ASD (N = 3064) to examine their financial well-being and its association with health expenditures for children. Extensive control variables (i.e., demographic and socioeconomic characteristics of children, respondents, and their families) are adjusted in analyses. Findings suggest that the amount of health expenditures is negatively associated with respondents' perception of their financial status. The significance of health expenditures disappears after household material hardship is adjusted. Health expenditures affect financial well-being mainly through resource competitions against family needs.

Dimensions of Caregiver Burden between Compound and Noncompound Caregivers of Adults with Autism.

The expanding population of adults with autism spectrum disorder (ASD) will increase the number of aging parents who often assume the role of primary caregivers. Among aging caregivers of adults with ASD, compound caregiving (caring for additional family members) has become an increasingly common scenario. However, limited research has examined the burden of compound caregivers and little is known about the specific dimensions of burden among this population. The present study aims to compare differences between compound 1, compound 2, and noncompound caregivers on specific dimensions of caregiver burden associated with adult children with ASD. Using a cross-sectional design, data were collected from 320 caregivers aged 50 and older through a web-based survey. Four dimensions of burden (time dependence, developmental, emotional, and financial) were compared using MANOVA. Compound 1 and noncompound caregivers experienced lower levels of time dependence burden than compound 2 caregivers. In terms of developmental burden, the social life of compound 2 caregivers was more affected by caregiving compared to noncompound caregivers. The findings have practice implications for identifying caregivers' multiple responsibilities and the multi-faceted impact of caregiving, and prioritizing interventions for compound caregivers and its subpopulations to reduce their time and developmental burden.

Mother-father physiological synchrony during conflict and moderation by parenting challenges: Findings from parents of children with autism spectrum disorder.

INTRODUCTION: Despite robust evidence in the broader family process literature for within-couple associations between romantic partners' physiological responding, this linkage has not been tested directly among parents raising a child with autism spectrum disorder (ASD). Explicit attention to these parents is warranted on the basis of the established family-wide challenges associated with the persistent emotional, functional, and behavior impairments typically observed in children with ASD (see Karst & Van Hecke, 2012). The first purpose of this study is to extend examination of within-couple associations between electrodermal activity (EDA) to mothers and fathers of children with ASD. The second purpose is to test 2 potential moderators of EDA synchrony reflecting heightened challenges among parents of children with ASD, namely parenting as an observed conflict topic and global parenting stress. METHOD: Both parents wore wrist sensors that captured continuous EDA during a recorded marital conflict interaction. Trained coders provided ratings of emotional intensity and conflict topics, including whether the topic of parenting was discussed. Parents reported their global level of parenting stress. Dyadic multilevel modeling was used to test our hypotheses and accounted for interaction factors (e.g., movement data, emotional intensity). RESULTS: Findings identified significant positive associations between parents' EDA. One moderator result emerged; specifically, father EDA was a stronger predictor of mother EDA when parenting was discussed as a conflict topic. CONCLUSION: This study extends evidence of within-couple physiological linkage to parents raising a child with ASD and provides a foundation for future research examining the family-wide implications of this linkage. (PsycInfo Database Record (c) 2021 APA, all rights reserved).

Burden of major depressive disorder and quality of life among mothers of children with autism spectrum disorder in urban bangladesh.

This cross-sectional study examined the burden of depression and quality of life (QoL) among mothers of children with autism spectrum disorder (ASD) attending six schools offering special educational services for children with ASD in urban Dhaka, Bangladesh. All consenting mothers were 18 years of age and older and met criteria for major depressive disorder (MDD) on the Structured Interview for DSM-V-TR Research Version-non patient edition administered by trained raters. QoL was assessed by the EuroQol five-dimensional standardized questionnaire. MDD was diagnosed in 45% of mothers and was proportionally higher among those who did not work outside the home, had no childcare support at home, expressed low level of satisfaction with the quality of providers when they had sought care for their children with ASD, and reported being recipient of negative attitudes from neighbors toward their children with ASD. QoL was negatively associated with MDD, mothers' illness, and low satisfaction with the health care providers for children with ASD, as well as experiencing a negative attitude by neighbors toward their children. QoL was positively associated with the reported family monthly income and improvement of the children with ASD on school attendance. The prevalence of MDD among mothers of children with ASD was high and associated with poor QoL. Integrating mental health services and supports for mothers in the ASD care of children is likely to address the high burden of depression they face, and improve their overall quality of life. Autism Res 2020, 13: 284-297. © 2019 International Society for Autism Research, Wiley Periodicals, Inc. LAY SUMMARY: The levels of depression were assessed among mothers of children with autism spectrum disorder (ASD) attending six schools offering special educational services for children with ASD in urban Dhaka, Bangladesh. Almost one in two mothers was found to have major depression. The quality of life of these mothers was generally poor. Integrating mental health services for mothers with ASD care in children is likely to address the burden of depression among the mothers of children with ASD and improve overall quality of life.

Intellectual disability and autism: socioeconomic impacts of informal caring, projected to 2030.

BACKGROUND: Intellectual disability and autism spectrum disorder (ASD) influence the interactions of a person with their environment and generate economic and socioeconomic costs for the person, their family and society. AIMS: To estimate costs of lost workforce participation due to informal caring for people with intellectual disability or autism spectrum disorders by estimating lost income to individuals, lost taxation payments to federal government and increased welfare payments. METHOD: We used a microsimulation model based on the Australian Bureau of Statistics' Surveys of Disability, Ageing and Carers (population surveys of people aged 15-64), and projected costs of caring from 2015 in 5-year intervals to 2030. RESULTS: The model estimated that informal carers of people with intellectual disability and/or ASD in Australia had aggregated lost income of AU$310 million, lost taxation of AU$100 million and increased welfare payments of AU$204 million in 2015. These are projected to increase to AU$432 million, AU$129 million and AU$254 million for income, taxation, and welfare respectively by 2030. The income gap of carers for people with intellectual disability and/or ASD is estimated to increase by 2030, meaning more financial stress for carers. CONCLUSIONS: Informal carers of people with intellectual disability and/or ASD experience significant loss of income, leading to increased welfare payments and reduced taxation revenue for governments; these are all projected to increase. Strategic policies supporting informal carers wishing to return to work could improve the financial and psychological impact of having a family member with intellectual disability and/or ASD. DECLARATION OF INTEREST: None.

Impact of multiple co-occurring emotional and behavioural conditions on children with autism and their families.

BACKGROUND: Comorbid conditions are very common in children and adults with autism spectrum disorder (ASD) and can affect school performance, adaptive skills and peer relationships. Comorbid conditions place strain on the family as well as the individual with ASD. This project aimed to determine the affect of comorbid conditions over and above child and family characteristics. METHODS: The present authors examined 3,055 cases of children with ASD who had varying numbers of comorbid conditions (i.e., ADHD, depression, anxiety or behaviour problems). RESULTS: Multiple comorbid conditions did have a unique impact on difficulties accessing services. A greater number of comorbid conditions impacted daily activity participation, but not a family's receipt of mental health care or respite, work changes or number of weekly hours dedicated to caring for a child with ASD. CONCLUSIONS: Families, practitioners, paraprofessionals and educators of children with autism should address comorbid conditions to ensure both child and family well-being.

Impact of Caregiver Burden on Quality of Life for Parents of Adult Children With Autism Spectrum Disorder.

This study sought to examine the impact of time, developmental, emotional, and financial burdens on the quality of life (QOL) for parents (aged 50+) of an adult child with autism spectrum disorder (ASD). Participants ( N = 320) completed a web-based survey and could indicate interest in participating in one-on-one follow up interviews. Multiple linear regression analysis indicated that two variables, developmental burden and impact of caregiving on finances, were statistically significant predictors of parents' QOL. Developmental burden was the strongest predictor of parental QOL, demonstrating an inverse relationship. Qualitatively, reported findings indicated that many parents were experiencing all four burdens. Findings highlighted the need to provide services and supports to alleviate burden among aging parents of adult children with ASD.

Psychological distress and perceived burden in caregivers of persons with autism spectrum disorder.

PURPOSE: To investigate psychological distress among parents of children with autism spectrum disorder and the associated sociodemographic factors influencing parents' distress. DESIGN AND METHODS: A cross-sectional design with a sample of 123 Jordanian parents providing care to children with autism spectrum disorder was used. A sociodemographic questionnaire, the Oberst Caregiving Burden Scale, the Bakas Caregiving Outcomes Scale, and the Hospital Anxiety and Depression Scale were utilized for data collection. FINDINGS: Parents reported moderate levels of burden, negative life changes, and borderline depression and anxiety. The perceived burden related to caregiving task difficulty positively correlated with that related to time spent on tasks and negatively correlated with caregivers' caregiving-related outcomes. The perceived burden of caregiving tasks was negatively correlated with depression and anxiety and anxiety levels were positively correlated with depression (P < 0.001). PRACTICE IMPLICATION: Healthcare providers, advanced practice nurses, and policy makers should be aware of the burden, anxiety, and depression experienced by caregivers of children with autism spectrum disorder.

Understanding the Family Impact of Autism Spectrum Disorder in a Racially and Ethnically Diverse Sample: Findings from the National Survey of Children with Special Health Care Needs.

Objectives Caregivers of children with autism spectrum disorder (ASD) experience stress at greater rates than caregivers of other children with developmental conditions. Little is known about how families from different racial and ethnic backgrounds report family impact beyond individual stressors associated with caregiving. This paper aims to examine differences in family impact variables among caregivers of ASD children from different racial/ethnic backgrounds. Methods Using data from the 2005-2006 and 2009-2010 National Survey of Children with Special Health Care Needs, this retrospective, cross-sectional study examined family impact among caregivers of children with ASD. Family impact was defined as financial impact, time spent caregiving, and work impact variables and evaluated in five racial/ethnicity groups: white, non-Hispanic; any race, English-speaking Hispanic; any race, Spanish-speaking Hispanic; black, non-Hispanic; and other race, non-Hispanic respondents (n = 5115). Multivariate logistic regression was used to analyze the association of race and ethnicity with family impact variables while controlling for child and family covariates. Results Significant differences were found between race/ethnicity groups of caregivers on financial spending of more than $500 per year on care and providing more than 11 h a week on direct child care. No significant differences were observed in job impact variables between race/ethnicity groups. Conclusions for Practice Racial/ethnic differences exist in providing and spending more on direct care, but they do not necessarily represent disparities. More research is needed to fully understand if family impact is affected by cultural differences in care provided for children with ASD.

The economic costs and its predictors for childhood autism spectrum disorders in Ireland: How is the burden distributed?

Autism spectrum disorders are associated with a substantial economic burden; although little is known about the relationship between state and family out-of-pocket expenditure. The objective of this study is to estimate the societal cost of childhood autism spectrum disorders and explain the variation in costs between state and family out-of-pocket expenditure. A bottom-up prevalence based cost-of-illness methodology was implemented using data from a combination of multiple convenience samples in Ireland of 195 parents of 222 children aged between 2 and 18 years of age with a clinically diagnosed autism spectrum disorder collected in 2014/2015. The findings show the average annual cost per child for families amounted to €28,464.89 related to private autism spectrum disorder services, lost income and informal care. By comparison, annual state expenditure per child on autism spectrum disorder-related health, social and educational resources was €14,192. Regression analyses indicate that autism spectrum disorder severity is significantly associated with higher out of pocket expenditures but not state health expenditures. The results suggest that parents are central to meeting the needs of young people with autism spectrum disorders in Ireland. Policy implications of these findings suggest that significant investment and commitment is needed to address the needs of individuals living with autism spectrum disorders and their families.

Burden of care perceived by the principal caregivers of autistic children and adolescents visiting health facilities in Lucknow City.

BACKGROUND: Autism is extraordinarily difficult for families to cope with for various reasons. Perceived burden and care for the child with autism, available forms of social support, and the interactions between the autistic child and other family members are areas of significant concern for families. OBJECTIVE: The objective of this study was to determine the burden of care perceived by the principal caregivers of autistic children or adolescent visiting health facilities in Lucknow city. METHODS: The cross-sectional study was conducted from October 2016 to September 2017 with a sample of 90 principal caregivers (aged <60 years) of autistic children and adolescents aged 3-19 years and diagnosed with autism, attending government and private health facilities providing treatment for autism in Lucknow, Uttar Pradesh, India. Validated tools were used to assess the burden of care and disability level of autistic children. Data were analyzed using the software SPSS version 16. RESULTS: The caregivers of autistic children and adolescents perceived the burden of care in different domains in varying extent with "caregiver's routine" and "taking responsibility" domains affected the most. The burden perceived was found to be affected by the permanent residence of caregiver, rural/urban dwelling, type of family, socioeconomic status, age at which diagnosis was made, knowledge about autism and the severity of autism. CONCLUSIONS: Availability and easy accessibility of autism treatment facilities must be the most probable reason for less burden perceived in three domains, by caregivers of urban settings and those belonging to Lucknow. Furthermore, better knowledge on autism and family and friends' support led to decrease in the burden perceived by the caregivers in various domains. Thus, it was found that the burden perceived can be reduced by universal availability of evidence-based early diagnosis and treatment of autism and improving the knowledge of caregivers about autism.

Perceived Burden in Caregivers of Children with Autism Spectrum Disorder.

BACKGROUND: Autism Spectrum Disorder is a lifelong developmental disorder that affects children and results in deficit in social interaction, communication and display of unusual pattern of behavior. Though caregiving is a normal parental duty, providing intensive care to a child with long-term problem is burdensome and impacts both physical and mental health of caregivers. The aim of this study was to obtain a picture of burden among caregivers raising children with Autism Spectrum Disorder. METHODS: We conducted a descriptive cross-sectional study to assess the burden of caregiving among sixty one parents of children with Autism Spectrum Disorder in the Kathmandu Valley, Nepal, using purposive sampling technique. The burden perceived by caregivers was assessed using standard tool Zarit Burden Interview-22. RESULTS: Average burden score was 41.49±12.25 which indicates that caregivers experienced moderate to severe range of burden. Level of education, anxiety and depression was found to be significantly associated with burden of caregiving. CONCLUSIONS: The study concluded that the burden of caregiving is most remarkably associated with emotional status and social life of the caregivers.

Understanding the Needs of Families of Children Who Are Deaf/Hard of Hearing with an Autism Spectrum Disorder.

BACKGROUND: There is a significant lack of evidence guiding our understanding of the needs of families of children who are deaf/hard of hearing (Deaf/HH) with an autism spectrum disorder (ASD). Much of our current knowledge is founded in case report studies with very small numbers of children with the dual diagnosis. PURPOSE: The purpose of this study was to gain an understanding of the factors relating to caregiver stress and needs (i.e., supports and interventions) in families of children who are Deaf/HH with ASD. RESEARCH DESIGN: Comparison groups of families of children who were Deaf/HH, families with a hearing child with ASD, and families of children who were Deaf/HH with ASD were administered standardized questionnaires of stress with brief qualitative questionnaires focusing on family-identified needs. STUDY SAMPLE: Six families of children with the dual diagnosis, four families of children who were Deaf/HH, and three families of children with ASD. DATA COLLECTION AND ANALYSIS: Surveys included demographic and support questionnaires, the Parenting Stress Index (PSI), the Pediatric Hearing Impairment Caregiver Experience, and a qualitative questionnaire. RESULTS: Families of children who were Deaf/HH with ASD had a higher median total stress score on the PSI as compared to families of children who were Deaf/HH only (58.5 versus 41.5, respectively; p = 0.02) and higher Child Domain scores (60 versus 43, respectively; p = 0.02), indicating higher levels of stress in families of children with the dual diagnosis. The families of children who were Deaf/HH with ASD reported similar levels of stress as families of children with ASD. CONCLUSIONS: Families of children who are Deaf/HH with an ASD experience stress and describe similar needs and priorities as families of hearing children with ASD. This suggests the needs related to having an autism spectrum disorder are of high priority in families of children with the dual diagnosis.

The impact on the family of the co-existing conditions of children with autism spectrum disorder.

We aimed to investigate whether the impact on families of children with Autism Spectrum Disorder (ASD) is associated with the number and/or type of emotional and behavioral co-existing conditions that parents/carers of children with ASD reported as occurring frequently. In addition, we examined whether there was a greater impact on families if their child was male, had lower levels of language, had more severe autism symptomatology, and whether impact was associated with the number and/or type of co-existing conditions. Families were recruited from large UK research databases. 420 parents/carers of children aged 3 years 2 months to 18 years 8 months completed the revised Impact on Family (IoF) Scale and reported on the frequency/rate of their child's co-existing conditions. Parents/carers reported higher mean IoF scores if their child: had a greater number of frequent co-existing conditions; had sleep problems; was only able to communicate physically; and had more severe autism symptomatology. The development and implementation of targeted treatment and management approaches are needed to reduce the impact of co-existing conditions on family life. Autism Res 2018, 11: 776-787. © 2018 International Society for Autism Research, Wiley Periodicals, Inc. LAY SUMMARY: Autism Spectrum Disorder (ASD) is commonly associated with emotional and/or behavior conditions that affect family life. Parents/carers of children with ASD who: (a) reported a greater number of frequent co-existing conditions, (b) had sleep problems, (c) were only able to communicate physically, and (d) had more severe symptoms characteristic of autism, reported a greater burden/strain on the family. Treatment approaches to target co-existing conditions alongside characteristics of ASD are needed to reduce their impact on family life.

Subjective Burden and Depression in Mothers of Children with Autism Spectrum Disorder in India: Moderating Effect of Social Support.

The quantitative study assessed subjective burden, depression, and the moderating effect of social support in mothers of children with autism spectrum disorder (ASD) in India. Seventy mothers were interviewed using a structured interview schedule, which measured their subjective burden, depression, and social support from family, friends, and significant others. Data was analyzed using descriptive statistics and multiple regression analysis with interaction terms. Half of the mothers in the study reported depression of clinical significance. Higher subjective burden significantly predicted higher depression. Of the three sources of support, only medium/high family support had a direct impact on depression and also moderated the impact of the subjective burden of depression. Implications for practice and policy are discussed.

The caregivers' perspectives of burden before and after an internet-based intervention of young persons with ADHD or autism spectrum disorder.

BACKGROUND: Everyday life in a family with a young person with attention-deficit/hyperactivity disorder (ADHD) and/or autism spectrum disorder (ASD), is multifaceted and may be a burden to the caregivers. The aim was to explore experiences of the caregiver situation, and how they were affected when the young persons received Internet-Based Support and Coaching (IBSC). METHOD: A multiple descriptive case design was used. Ten caregivers of 10 young persons with ADHD and/or ASD participated, and completed a questionnaire, measuring caregiver burden. Three analysis methods were used; statistical analysis of the caregivers' scores, case reports and qualitative content analysis of an open question. RESULTS: Most of the caregivers never had to provide hands-on help with primary ADL, and were able to cope with the young person's problems. The caregivers' role required patience. They had major worries about the future, when they would no longer be around to provide support. After the intervention, there was no common trajectory for all caregivers; their situation either remained unchanged, improved or deteriorated. CONCLUSIONS: The caregiver's burden is more or less complex when the young person undergoes the transition into adulthood. The caregiver burden decreased in some families following the young person's participation in IBSC.

Design and Cohort Characteristics of the Social Spectrum Study: A Multicenter Study of the Autism Spectrum Among Clinically Referred Children.

This paper provides an overview of the design and cohort characteristics of the Social Spectrum Study: a clinical cohort study that used a two-phase sampling design to identify children at risk for ASD. After screening 1281 children aged 2.5-10 years who had been consecutively referred to one of six mental health services in the Netherlands, children who screened positive for ASD (n = 428) and a random selection of screen negatives (n = 240) were invited to participate in diagnostic assessments and questionnaires regarding the child, family and society. A 1-year follow-up was also conducted. Results from this study may contribute to knowledge of the identification and characterization of children with ASD, family processes, and the impact of ASD on the family and society.

Infantile autism: impact of diagnosis and repercussions in family relationships. / Autismo infantil: impacto do diagnóstico e repercussões nas relações familiares.

OBJECTIVE: To analyse the context in which the diagnosis of autism is revealed and the impact of this revelation on family relationships. METHODS: This is a qualitative study with 10 families of autistic children assisted at the Psychosocial Care Centre for Children and Youths in a municipality of Paraíba. Data were collected between July and August 2013 through semi-structured interviews and interpreted using thematic content analysis. RESULTS: The identified Main Unit of Analysis and the respective categories were: the impact of disclosing the diagnosis of autism to the family; characteristics of diagnosis disclosure: the place, time, and the dialogic relationship between the professional and the family; changes in family relationships and the mother's burden when caring for the autistic child. CONCLUSIONS: The health professionals who report autism should better prepare the family to cope with the difficulties of this syndrome and autonomously care for the autistic child.

Disease Burden and Symptom Structure of Autism in Neurofibromatosis Type 1: A Study of the International NF1-ASD Consortium Team (INFACT).

IMPORTANCE: Recent reports have demonstrated a higher incidence of autism spectrum disorder (ASD) and substantially elevated autistic trait burden in individuals with neurofibromatosis type 1 (NF1). However, important discrepancies regarding the distribution of autistic traits, sex predominance, and association between ASD symptoms and attentional problems have emerged, and critical features of the ASD phenotype within NF1 have never been adequately explored. Establishing NF1 as a monogenic cause for ASD has important implications for affected patients and for future research focused on establishing convergent pathogenic mechanisms relevant to the potential treatment targets for ASD. OBJECTIVE: To characterize the quantitative autistic trait (QAT) burden in a pooled NF1 data set. DESIGN, SETTING, AND PARTICIPANTS: Anonymized, individual-level primary data were accumulated from 6 tertiary referral centers in the United States, Belgium, United Kingdom, and Australia. A total of 531 individuals recruited from NF1 clinical centers were included in the study. MAIN OUTCOMES AND MEASURES: Distribution of ASD traits (Social Responsiveness Scale, second edition [SRS-2], with T scores of ≥75 associated with a categorical ASD diagnosis); attention-deficit/hyperactivity disorder (ADHD) traits (4 versions of Conners Rating Scale, with T scores of ≥65 indicating clinically significant ADHD symptoms); ASD symptom structure, latent structure, base rate derived from mixture modeling; and familiality. RESULTS: Of the 531 patients included in the analysis, 247 were male (46.5%); median age was 11 years (range, 2.5-83.9 years). QAT scores were continuously distributed and pathologically shifted; 13.2% (95% CI, 10.3%-16.1%) of individuals scored within the most severe range (ie, above the first percentile of the general population distribution) in which the male to female ratio was markedly attenuated (1.6:1) relative to idiopathic ASD. Autistic symptoms in this NF1 cohort demonstrated a robust unitary factor structure, with the first principal component explaining 30.9% of the variance in SRS-2 scores, and a strong association with ADHD symptoms (r = 0.61). Within-family correlation for QAT burden (intraclass correlation coefficient, 0.73 in NF1-affected first-degree relatives) exceeded that observed in the general population and ASD family samples. CONCLUSIONS AND RELEVANCE: This study provides confirmation that the diversity of mutations that give rise to NF1 function as quantitative trait loci for ASD. Moreover, the within-family correlation implicates a high degree of mutational specificity for this associated phenotype. Clinicians should be alerted to the increased frequency of this disabling comorbidity, and the scientific community should be aware of the potential for this monogenic disorder to help elucidate the biological features of idiopathic autism.