Analyzing the Quality of Life in Individuals with Fragile X Syndrome in Relation to Sleep and Mental Health.

The purpose of this paper was to examine the physical, emotional, social and school functioning domains of quality of life of individuals with Fragile X Syndrome, in relation to mental health and sleep patterns to gain a better understanding of how these aspects are affected by the disorder. This study included 119 individuals with Fragile X Syndrome who were given different cognitive examinations by a neuropsychologist or by parent-proxy questionnaires. This study focused on the Pediatric Quality of Life Inventory (PedsQoL), the Anxiety, Depression and Mood Scale (ADAMS), the Children's Sleep Habits Questionnaire (CSHQ), but did include other cognitive tests (Vineland Adaptive Behaviour Scales, Nonverbal IQ, Autism Diagnostic Observation Schedule). We identified significant associations between decreases in emotional, social and school domains of PedsQoL and the ADAMS subtests of Generalized Anxiety, Manic/Hyperactivity and Obsessive/Compulsivity, with the subtest of Depressed Mood having associations with lower physical and emotional domains. We also identified a significant impact between CSHQ subtests of Sleep Anxiety, Night Wakings, Daytime Sleepiness, and Parasomnia with the emotional and school domains of PedsQoL. There were associations connecting school functioning with Bedtime Resistance, and additional associations connecting emotional functioning with Sleep Duration and Sleep Onset Delay. Physical functioning was also associated with Sleep Anxiety. Our study shows how mental health and sleep defects impact improper sleep patterns and mental health which leads to decreases in the quality of life for individuals with FXS, and how it is important to screen for these symptoms in order to alleviate issues.

Measure what matters: considerations for outcome measurement of care coordination for children with neurodevelopmental disabilities and medical complexity.

Introduction: Care Coordination (CC) is a significant intervention to enhance family's capacity in caring for children with neurodevelopmental disability and medical complexity (NDD-MC). CC assists with integration of medical and behavioral care and services, partnerships with medical and community-based supports, and access to medical, behavioral, and educational supports and services. Although there is some consensus on the principles that characterize optimal CC for children with NDD-MC, challenges remain in measuring and quantifying the impacts of CC related to these principles. Two key challenges include: (1) identification of measures that capture CC impacts from the medical system, care provider, and family perspectives; and (2) recognition of the important community context outside of a hospital or clinical setting. Methods: This study used a multilevel model variant of the triangulation mixed methods design to assess the impact of a CC project implemented in Alberta, Canada, on family quality of life, resource use, and care integration at the broader environmental and household levels. At the broader environmental level, we used linked administrative data. At the household level we used quantitative pre-post survey datasets, and aggregate findings from qualitative interviews to measure group-level impacts and an embedded multiple-case design to draw comparisons, capture the nuances of children with NDD-MC and their families, and expand on factors driving the high variability in outcome measures. Three theoretical propositions formed the basis of the analytical strategy for our case study evidence to explore factors affecting the high variability in outcome measures. Discussion: This study expanded on the factors used to measure the outcomes of CC and adds to our understanding of how CC as an intervention impacts resource use, quality of life, and care integration of children with NDD-MC and their families. Given the heterogeneous nature of this population, evaluation studies that account for the variable and multi-level impacts of CC interventions are critical to inform practice, implementation, and policy of CC for children with NDD-MC.

Children with Disabilities in Canada during the COVID-19 Pandemic: An Analysis of COVID-19 Policies through a Disability Rights Lens.

Children with disabilities were especially vulnerable during the COVID-19 pandemic, and policies designed to mitigate its effects were limited in addressing their needs. We analyzed Canadian policies related to children with disabilities and their families during the COVID-19 pandemic to identify the extent to which these policies aligned with the United Nations Convention on the Rights of Persons with Disabilities (UN CRPD) and responded to their mental health needs by conducting a systematic collection of Canadian provincial/territorial policies produced during the pandemic, building a categorization dictionary based on the UN CRPD, using text mining, and thematic analysis to identify policies' alignment with the UN CRPD and mental health supports. Mental health was addressed as a factor of importance in many policy documents, but specific interventions to promote or treat mental health were scarce. Most public health policies and recommendations are related to educational settings, demonstrating how public health for children with disabilities relies on education and community that may be out of the healthcare system and unavailable during extended periods of the pandemic. Policies often acknowledged the challenges faced by children with disabilities and their families but offered few mitigation strategies with limited considerations for human rights protection.

Pan-Canadian caregiver experiences in accessing government disability programs: A mixed methods study.

BACKGROUND: At present, little is known about the factors that contribute to the relatively low uptake of government-funded disability programs in Canada. AIM: Understand how parents/caregivers of Canadian youth with neurodevelopmental disability (NDD) experience the process of applying for and accessing disability programs. METHODS AND PROCEDURES: This mixed methods sequential explanatory study utilized two phases: an online survey (quantitative), followed by semi-structured interviews (qualitative). The quantitative phase gathered sociodemographic information and preliminary information about participant experiences applying for and accessing programs. The qualitative phase provided greater depth by asking participants to describe barriers and facilitators to program access. OUTCOMES AND RESULTS: 499 participants completed the online survey and 81 participants completed an interview. Analysis of survey data revealed that many participants are not accessing disability programs and experience difficulty when applying. Regression analyses revealed that factors relating to the process of applying and applicant/family attributes are significantly associated with program access. Inductive thematic analysis of interview data revealed four barriers and three facilitators to access. Integration of findings provided an overview of the multi-faceted journey to program access. CONCLUSIONS AND IMPLICATIONS: The results of this study highlight policy changes that are needed to ensure disability programs adequately support Canadian families.

Impacts of the COVID-19 Pandemic: Pan-Canadian Perspectives From Parents and Caregivers of Youth With Neurodevelopmental Disabilities.

INTRODUCTION: The COVID-19 pandemic brought unprecedented challenges for youth with neurodevelopmental disabilities (NDD) and their families. Although health measures were implemented to contain the COVID-19 virus, they disrupted public service, profoundly impacting youth and their families' access to services. This study sought to better understand the perspectives and experiences of parents and caregivers of youth with NDD across Canada in accessing services and their mental health needs during the pandemic. METHOD: The study used a qualitative research design in which we interviewed 40 parents and caregivers across Canada. RESULTS: The results enabled us to understand the impact of service disruptions in significant areas of life, including health, education, employment, and risk mitigation. DISCUSSION: Policymakers must consider a disability-inclusive lens during public health emergency planning and response to reduce the disproportionate impacts faced by youth with NDD and their families.

Mental health challenges during COVID-19: perspectives from parents with children with neurodevelopmental disabilities.

BACKGROUND: The global pandemic and subsequent denials, delays, and disruptions in essential daily activities created significant challenges for children with neurodevelopmental disorders (NDDs) and their parents. Public health restrictions during the COVID-19 pandemic limited access to supports and services required by children with NDDs to maintain their health and well-being. OBJECTIVE: This study sought to understand the impacts of these public health measures and restrictions on mental health from the perspective of parents with children with NDDs to inform pathways for public health policies responsive to the needs of this population. METHOD: Interpretive descriptive design was used to guide data collection and data analysis. Forty caregivers were interviewed about their experience with pandemic restrictions. FINDINGS: Generic policy measures contributed to many gaps in families' social support systems and contributed to mental health challenges for children and their parents. Four themes emerged: 1) lack of social networks and activities, 2) lack of access to health and social supports, 3) tension in the family unit, and 4) impact on mental health for children and their parents. RECOMMENDATIONS: Emergency preparedness planning requires a disability inclusive approach allocating resources for family supports in the home and community. Families identified supports to minimize further pandemic disruptions and enhance recovery.

Generic preference-based health-related quality of life in children with neurodevelopmental disorders: a scoping review.

AIM: To describe how generic preference-based health-related quality of life (HRQoL) instruments have been used in research involving children with neurodevelopmental disorders (NDD). METHOD: A systematic search of nine databases identified studies that used generic preference-based HRQoL instruments in children with NDD. Data extracted following the Preferred Reporting Items for Systematic Review and Meta-Analyses extension for Scoping Review guidelines included type of NDD, instrument used, respondent type, justification, and critical appraisal for these selections. RESULTS: Thirty-six studies were identified: four cost-utility analyses; 15 HRQoL assessments; five economic burden studies; three intervention studies; and nine 'other'. The Health Utilities Index (Mark 2 and Mark 3) and EuroQoL 5D (EQ-5D; three-level EQ-5D, five-level EQ-5D, and the youth version of the EQ-5D) instruments were most frequently used (44% and 31% respectively). The relatively low use of these instruments overall may be due to a lack of psychometric evidence, inconsistency in justification for and lack of clarity on appropriate respondent type and age, and geographical challenges in applying preference weights. INTERPRETATION: This study highlights the dearth of studies using generic preference-based HRQoL instruments in children with NDD. The use of cost-utility analysis in this field is limited and validation of these instruments for children with NDD is needed. The quality of data should be considered before guiding policy and care decisions. WHAT THIS PAPER ADDS: Limited use of generic preference-based health-related quality of life (HRQoL) instruments in studies on children with neurodevelopmental disorders. Only 11% of studies were cost-utility analyses. Inconsistencies in justification for choosing generic preference-based HRQoL instruments and respondent types.