Children's Health-related Quality Research Articles

Religious Belief Among Women in Australia: Characteristics and Role in Influencing Children's Health-Related Quality of Life and Lifestyle.

Religiosity can be an important factor in women's health-related behaviour, attitudes, and decision-making. Evidence however, regarding the religiosity of mothers and its influence on child health, is scarce. Based on a large population-based cohort in Australia, we aim to examine the religiosity of women in Australia and the association of maternal religiosity with children's health-related quality of life (HRQOL) and lifestyle. Our findings indicate that (1) maternal religious involvement was higher for women with higher education levels, ascertained religious values in decision-making, and abstinence from binge drinking in the household, (2) maternal religiosity positively influenced their children's HRQOL, (3) children of mothers who were more religious had less worries or fewer school-work problems, but the children of mothers with stronger religious beliefs used more internet/computer during the week but had less time playing games on weekends. This study provides additional specificity to inform future health interventions in religious community contexts to enhance the positive influence of maternal religious belief for better development of their children.

A multicenter randomized trial to compare automatic versus as-needed follow-up for children hospitalized with common infections: The FAAN-C trial protocol.

Physicians commonly recommend automatic primary care follow-up visits to children being discharged from the hospital. While automatic follow-up provides an opportunity to address postdischarge needs, the alternative is as-needed follow-up. With this strategy, families monitor their child's symptoms and decide if they need a follow-up visit in the days after discharge. In addition to being family centered, as-needed follow-up has the potential to reduce time and financial burdens on both families and the healthcare system. As-needed follow-up has been shown to be safe and effective for children hospitalized with bronchiolitis, but the extent to which hospitalized children with other common conditions might benefit from as-needed follow-up is unclear. The Follow-up Automatically versus As-Needed Comparison (FAAN-C, or "fancy") trial is a multicenter randomized controlled trial. Children who are hospitalized for pneumonia, urinary tract infection, skin and soft tissue infection, or acute gastroenteritis are eligible to participate. Participants are randomized to an as-needed versus automatic posthospitalization follow-up recommendation. The sample size estimate is 2674 participants and the primary outcome is all-cause hospital readmission within 14 days of discharge. Secondary outcomes are medical interventions and child health-related quality of life. Analyses will be conducted in an intention-to-treat manner, testing noninferiority of as-needed follow-up compared with automatic follow-up. FAAN-C will elucidate the relative benefits of an as-needed versus automatic follow-up recommendation, informing one of the most common decisions faced by families of hospitalized children and their medical providers. Findings from FAAN-C will also have implications for national quality metrics and guidelines.

The effect of 3D modeling on family quality of life, surgical success, and patient outcomes in congenital heart diseases: objectives and design of a randomized controlled trial.

Understanding the severity of the disease from the parents' perspective can lead to better patient outcomes, improving both the child's health-related quality of life and the family's quality of life. The implementation of 3-dimensional (3D) modeling technology in care is critical from a translational science perspective. The purpose of this study is to determine the effect of 3D modeling on family quality of life, surgical success, and patient outcomes in congenital heart diseases. Additionally, we aim to identify challenges and potential solutions related to this innovative technology. The study is a two-group pretest-posttest randomized controlled trial protocol. The sample size is 15 in the experimental group and 15 in the control group. The experimental group's heart models will be made from their own computed tomography (CT) images and printed using a 3D printer. The experimental group will receive surgical simulation and preoperative parent education with their 3D heart model. The control group will receive the same parent education using the standard anatomical model. Both groups will complete the Sociodemographic Information Form, the Surgical Simulation Evaluation Form - Part I-II, and the Pediatric Quality of Life Inventory (PedsQL) Family Impacts Module. The primary outcome of the research is the average PedsQL Family Impacts Module score. Secondary outcome measurement includes surgical success and patient outcomes. Separate analyses will be conducted for each outcome and compared between the intervention and control groups. Anomalies that can be clearly understood by parents according to the actual size and dimensions of the child's heart will affect the preoperative preparation of the surgical procedure and the recovery rate in the postoperative period.

Differential associations between Quantity, content, and context of screen time, and Children's health-related quality of life: A two-wave study

Existing literature suggests that lengthy durations of screen time (ST) are associated with lower levels of health-related quality of life (HRQoL) and other negative outcomes in children. Nonetheless, the current available evidence largely relies on cross-sectional studies. In addition, data on the content and context (parent–child co-use and solitary use) of ST are scarce. We set out to conduct a one-year, two-wave longitudinal study to investigate the association between exposure to different types of ST and HRQoL among primary school children in Hong Kong, from 2021 to 2022. A population representative random sample (N = 1428) participated in the study. Results from pooled ordinary least squares regressions showed children's ST duration was significantly associated with worse HRQoL (β = −1.101, p < 0.0001). However, interactions of ST content and context, including parent–child co-use × TV viewing (β = 1.305, p < 0.0001), parent–child co-use × video gaming (β = 0.280, p < 0.0001), solitary use × video gaming (β = 0.198, p < 0.0001), and solitary use × social media use (β = 0.454, p < 0.0001) were significantly associated with better HRQoL. Our findings suggest that increased overall durations of ST are associated with worse HRQoL in children, but this association is impacted by the types of ST content and context.

Associations among COVID-19 Family Stress, Family Functioning, and Child Health-Related Quality of Life through Lifestyle Behaviors in Children.

The COVID-19 pandemic has resulted in lasting effects on children, necessitating a thorough understanding of its impact for effective recovery planning. This study investigated the associations among COVID-19 family stress, family functioning, children's lifestyle behaviors (i.e., healthy food intake, unhealthy food intake, physical activity, and screen time), and their health-related quality of life (HRQOL). Data from a 2022 survey of parents with children aged 5 to 12 (mean age of boys: 8.36, mean age of girls: 7.76) in the United States through the online Prolific platform were analyzed using path analysis and gender-based multi-group analysis. The results showed an inverse relationship between family stressors and functioning (β = -0.39, p < 0.05). COVID-19 family stress was negatively related to child physical HRQOL (β = -0.20, p < 0.05) but not psychosocial HRQOL. Family functioning showed a positive relation with child healthy food intake (β = 0.26, p < 0.05) and a negative relation with unhealthy diet consumption (β = -0.27, p < 0.05), while no significant associations were found with child physical activity and screen time. Family functioning was indirectly associated with both types of HRQOL through the child's eating patterns. These relationships were more pronounced for girls. The findings point to a complex interplay between family stress and functioning, dietary habits, and the HRQOL of children during the COVID-19 pandemic, particularly concerning girls' food intake and well-being.

Children's and parents' experiences of home care provided by hospital staff: A scoping review.

To describe what is known from existing scientific literature on children's and parents' experiences of hospital-based home care and to identify future research areas. The scoping review design used adheres to the methodological framework of Arksey and O'Malley, and to the PRISMA-ScR checklist. A systematic search was conducted, and peer-reviewed scientific papers were screened through the application of Rayyan software. Data were extracted and presented in table and synthesised thematically as narrative text. Searches were carried out November 2021 and updated November 2022 in the CINAHL, MEDLINE, Embase, Cochrane, Scopus, Web of Science, Academic Search Elite, and Amed databases and Google Scholar. A total of 1950 studies were screened and assessed for eligibility. Eight studies met the inclusion criteria by reporting on parents' experiences, whereas five out of these eight studies also reported on the experiences of children. Parents of children with cancer and preterm children reported feeling more in control, being empowered, and being more connected to their children's care team when their children were receiving hospital-based home care. The family's own resources were activated, and they felt more involved in their children's care compared to being in an inpatient setting. Children with cancer, acute infection, chronic disease, and/or a syndrome reported feeling safer and more comfortable in their home environment and experienced better interaction with their care providers. Some aspects of the children's health-related quality of life (HRQOL) were improved. The identified studies indicate that hospital-based home care is a valued alternative to traditional inpatient care by both parents and children. The mode of care has no crucial negative effects. Future studies should encompass the experiences of children with different diagnoses and syndromes and compare patients treated in a traditional hospital setting with those in a hospital-based home-care programme. Children's and parents' experiences of HBHC indicate that it offers a good solution if parents are well prepared and feel in control. In addition, certain structural conditions must be in place before this type of care can be established: there must be a certain number of patients and the hospital must not be too far away. In the field of neonatal home care, professionals should be more responsive to fathers' needs and tailor support by focusing on their individual experiences and needs. Our findings may guide and inform best practice for present and future providers of HBHC. Hospital-based home care can offer families a greater degree of autonomy, more flexible care options, improved family functioning, improved communication with care providers, and more control over the child's care. When certain structural conditions are met, such as a certain number of patients, the hospital not being too far away, and parents being well prepared and feeling in control, then hospital-based home care is valued as an alternative to traditional inpatient care. Specific aspects of children's HRQOL may improve, and the psychosocial burden on the family does not increase. HBHC provides a valued alternative to traditional inpatient care and allows families to receive care in the comfort of their own home. Our findings may guide and inform best practice for present and future providers of hospital-based home care. In this scoping review, we have adhered to the Preferred Reporting Items for Systematic reviews and Meta Analyses Extension for Scoping Reviews checklist. There has been no direct patient or public contribution to the review. Not required.

Trajectory of health-related quality of life in parents of children treated with epilepsy surgery versus medical therapy.

Child health-related quality of life (HRQOL) has been shown to improve after epilepsy surgery and is linked to parent HRQOL. We postulated that the HRQOL of parents whose children underwent epilepsy surgery would improve over two years compared to those treated with medical therapy. The aim of the study was to evaluate the trajectory of HRQOL of parents whose children received treatment with epilepsy surgery or medical therapy over two years. This multi-center study recruited parents whose children were evaluated for epilepsy surgery. Parents completed measures of care-related QOL (CarerQOL) at the time of their children's surgical evaluation, 6 months, 1 year, and 2 years later. Additional measures included parent anxiety and depression, satisfaction with family relationships, family resources and demands, and child clinical variables. A linear mixed model was used to compare the trajectories of parent HRQOL of surgical and medical patients, adjusting for baseline clinical, parent, and family characteristics. There were 111 children treated with surgery and 154 with medical therapy. The trajectory of parent HRQOL was similar among parents of surgical and medical patients over the two-year follow-up. However, HRQOL of parents of surgical patients was 3.0 points higher (95%CI -0.1, 6.1) across the follow-up period compared to parents of medical patients. Parents of seizure-free children reported 2.3 points (95%CI 0.2, 4.4) higher HRQOL relative to parents of non-seizure-free children across the two-year follow-up. Parent HRQOL did not improve after their children were treated with epilepsy surgery, possibly related to ongoing comorbidities in children.

The RETRIEVE Checklist for Studies Reporting the Elicitation of Stated Preferences for Child Health-Related Quality of Life.

Recent systematic reviews show varying methods for eliciting, modelling, and reporting preference-based values for child health-related quality-of-life (HRQoL) outcomes, thus producing valuesets with different characteristics. Reporting in many of the reviewed studies was found to be incomplete and inconsistent, making them difficult to assess. Checklists can help to improve standards of reporting; however, existing checklists do not address methodological issues for valuing child HRQoL.Existing checklists also focus on reporting methods and processes used in developing HRQoL values, with less focus on reporting of the values' key characteristics and properties. We aimed to develop a checklist for studies generating values for child HRQoL, including for disease-specific states and value sets for generic child HRQoL instruments. A conceptual model provided a structure for grouping items into five modules. Potential items were sourced from an adult HRQoL checklist review, with additional items specific to children developed using recent reviews. Checklist items were reduced by eliminating duplication and overlap, then refined for relevance and clarity via an iterative process. Long and short checklist versions were produced for different user needs. The resulting long RETRIEVE contains 83 items, with modules for reporting methods (A-D) and characteristics of values (E), for researchers planning and reporting child health valuation studies. The short RETRIEVE contains 14 items for decision makers or researchers choosing value sets. Applying the RETRIEVE checklists to relevant studies suggests feasibility. RETRIEVE has the potential to improve completeness in the reporting of preference-based values for child HRQOL outcomes and to improve assessment of preference-based value sets.

Understanding UK public views on normative decisions made to value health-related quality of life in children: A qualitative study

Developing methodology for measuring and valuing child health-related quality of life (HRQoL) is a priority for health technology agencies. One aspect of this is normative decisions that are made in child HRQoL valuation. This qualitative study aimed to better understand adult public opinion on the normative questions of whose preferences to elicit (adults, children, or both) and from which perspective (who should be imagined living in impaired health), when valuing child HRQoL. Opinions of the adult UK public (N = 32) were solicited using online semi-structured focus groups, featuring a breadth of age, sex, ethnicities, and responsibility for children under 18 years. Participants were provided with bespoke informational material on health state valuation and were probed for their views. Arguments for and against different positions were discussed. Data was analysed using framework analysis. Participants demonstrated near-to-universal agreement that children should be involved in valuation in some form, yet this should differ depending on age or maturity. There was strong support for approaches combining involvement from children and adults (e.g., their parents), especially for younger children. There was little intuitive support for the ‘taxpayer argument’ for asking taxpaying adults. In the context of greater involvement of children in valuation, most participants supported using an ‘own’ perspective. Most participants thought that valuation study participants should know the exercise is about valuing child health states for ethical reasons. Informed views from the UK public on who should be asked and with what perspective when valuing child HRQoL appear to differ from normative positions previously advocated by some health economists, such as prioritising the preferences of taxpaying adults. In contrast, the results suggest including adults and children in valuation, with the proviso that the children are of an appropriate age and level of maturity, and that an own perspective is used wherever possible.

Child characteristics associated with child quality of life and parenting stress in Angelman syndrome.

Angelman syndrome (AS) is a rare neurodevelopmental disorder characterised by severe intellectual disability, movement disorder, epilepsy, sleeping problems, and behavioural issues. Little is known on child health-related quality of life (HRQoL) in AS. AS family studies have reported elevated parenting stress and a high impact of the child's syndrome on the parent. It is unclear which factors influence child HRQoL and parenting stress/impact in AS. We collected data prospectively through standardised clinical assessments of children with AS at the ENCORE Expertise centre for Angelman Syndrome at the Erasmus MC Sophia Children's Hospital. A linear regression analysis was conducted for the following outcome variables: (1) child HRQoL (Infant and Toddler Quality of Life Questionnaire); (2) the impact of the child's syndrome on the parent (Infant and Toddler Quality of Life Questionnaire); and (3) parenting stress (Parenting Stress Index). Predictor variables were child genotype, epilepsy, sleeping problems (Sleep Disturbance Scale for Children), cognitive developmental level (Bayley Cognition Scale), autistic features (Autism Diagnostic Observation Schedule) and emotional/behavioural problems (Child Behaviour Checklist). Covariates were sex, age and socio-economic status. The study sample consisted of 73 children with AS, mean age=9.1years, range=2-18years. Emotional/behavioural problems were the strongest significant predictor of lowered child HRQoL. Internalising problems were driving this effect. In addition, having the deletion genotype and higher age was related to lower child HRQoL. Sleeping problems were related to a higher impact of the child's syndrome on the parent. Finally, emotional/behavioural problems were associated with higher parenting stress. Cognitive developmental level, autistic features and epilepsy were not a significant predictor of child HRQoL and parenting stress/impact. These results suggest that interventions aimed at increasing child HRQoL and decreasing parenting stress/impact in AS should focus on child emotional/behavioural problems and sleeping problems, using a family-centred approach.

Potential Mediating Roles of Children's Health-Related Quality of Life and Weight-Related Behaviors in the Relationship Between Socio-Educational Advantage and Weight Status.

Background: Socioeconomic status is inversely associated with weight status in developed countries. Underlying mechanisms are still to be clarified. Our aim was to determine if weight-related behaviors and health-related quality of life (HRQoL) mediate the relationship between socio-educational advantage and weight status in Australian children 9 to 13 years of age. Methods: Secondary analysis of data collected by two cluster randomized trials (2019 wave). We measured children's (n = 3978) height, weight, diet, physical activity, sedentary behavior, and HRQoL. Socio-.educational advantage was assessed at school level using the Index of Community Socio-Educational Advantage (ICSEA). A counterfactual framework was used to explore potential mediators in the relationship between socio-educational disadvantage and (1) overweight/obesity and (2) BMI z-score (BMIz). Results: Low socio-educational advantage was associated with increased overweight/obesity and BMIz. The overweight/obesity association was mediated by sedentary behavior (natural indirect effect as a proportion of total, 7.5%) sugar-sweetened beverage (SSB) consumption (12.7%), physical functioning (11.9%), psychosocial health (10.9%), school (6.8%) and social functioning (15.6%), and total HRQoL score (13.8%). The ICSEA-BMIz relationship was mediated by sedentary behavior (5.7%), sleep duration (4.1%), SSB (10.6%), physical functioning (9.9%), psychosocial health (9.1%), school (5.5%) and social (13.7%) functioning, and total HRQoL score (11.7%). Conclusion: Victorian Children with low socio-educational advantage were more likely to be living with overweight or obesity. This relationship was mediated by children's sedentary behavior, SSB consumption, and HRQoL. Trial Registration: Australian New Zealand Clinical Trials Registry (ANZCTR): ACTRN12616000980437 (registered July 26, 2016, retrospectively registered) and ACTRN12618001986268 (registered December 11, 2018, prospectively registered). https://www.anzctr.org.au/Trial/Registration.

The Burden of Sickle Cell Disease on Children and Their Caregivers: Caregiver Reports of Children's Health-Related Quality of Life and School Experiences, Caregiver Burden, and Their Association with Frequency of Vaso-Occlusive Crises.

Children with sickle cell disease (SCD) experience a multiplex of disease-related symptoms and complications, including vaso-occlusive crises (VOCs), episodes characterized by extreme pain. A cross-sectional observational survey examined the health-related quality of life (HRQoL) and school experiences of children with SCD 2 months-11 years, burden experienced by their caregivers, and associations between these outcomes and VOC frequency. Caregivers (N=167) of children with SCD in the US completed the Infant-Toddler Quality of Life-Short Form 47 (ITQoL-SF47) for children 2 months-4 years, the Child Health Questionnaire-Parent Form 50 (CHQ-PF50) and PROMIS Pain Interference and Sleep Disturbance Parent Proxy short forms for children 5-11 years, and a study-specific survey of school experiences. Children with SCD 2 months-4 years had lower ITQoL-SF47 scores (ie, worse HRQoL, p<0.001) than a normative sample of children; across domains, differences ranged from 18.73-45.03 points and exceeded minimal important difference (MID) thresholds. Except for the behavior domain, children with SCD 5-11 years had lower scores on all CHQ-PF50 domains than the normative sample (p<0.001); differences ranged from 6.78-36.37 points and exceeded MID thresholds. Children with more frequent VOCs had lower HRQoL and worse school experiences than children with less frequent VOCs (p<0.05, except for behavior domains). The largest differences based on VOC frequency were observed for overall health and bodily pain/discomfort among children 2 months-4 years (differences=40.88 and 32.50 points, respectively), and bodily pain and role/social limitations due to physical health among children 5-11 years (differences=38.99 and 37.80, respectively). Caregivers of children with more frequent VOCs experienced greater burden than caregivers of children with less frequent VOCs, though specific areas of impact (eg, caregiver emotions, time) differed across child age groups. VOC frequency is negatively associated with HRQoL, highlighting the burden experienced by children with SCD and their caregivers.

Health-related quality of life in Chinese children and adolescents with cancer.

Cancer diagnosis and related treatment can have multiple impacts on children's health-related quality of life (HRQoL). This study aimed to determine profiles of HRQoL in Chinese children and adolescents with cancer based on patient-reported outcomes. This cross-sectional study recruited 310 pediatric patients with cancer (in treatment and survivorship) aged 8-17 years old and their family caregivers from four hospitals in China. The participants were asked to complete the PROMIS Pediatric-25 Profile 2.0, along with a demographic and clinical characteristics questionnaire, fatigue screening item, and an item assessing the family caregiver's coping style. Latent profile analysis was used to identify profiles. Two profiles were identified: Profile 1, "low symptom and high function" (n = 147, 47.4%); and Profile 2, "high symptom and low function" (n = 163, 52.5%). Children reporting ≥3 on the five-point Symptom Distress Scale fatigue screening item were more likely to be in Profile 2 (OR = 1.961; 95% CI: 1.098-3.501). Participants were less likely to be in Profile 2 if they were in survivorship (OR = 0.494; 95% CI: 0.271-0.903), or their caregiver's coping style was identified as facing positively (OR = 0.439; 95% CI: 0.274-0.703). The identified profiles demonstrate the heterogeneity in HRQoL among pediatric patients with cancer, and the importance of supporting caregivers' coping as a means of supporting the child. Knowledge of these profiles can assist clinicians in better identifying and targeting interventions for children with cancer.

Post-PICU sleep efficiency and quality of life in infants and toddlers with acquired brain injury.

We aimed to investigate the use of sleep efficiency (SE) as a measure of sleep disturbance in infants and toddlers with acquired brain injury (ABI) and evaluate associations between SE and child health-related quality of life and family outcomes. Retrospective cohort study of 101 children ages 3-36 months who survived critical care for ABI. SE was quantified from the Brief Infant Sleep Questionnaire as a ratio of nighttime sleep to total time in bed; poor SE was defined as < 80%. Outcome measures included the Pediatric Quality of Life Inventory Core Total Score (health-related quality of life) and Family Impact Module Total Score. Spearman's correlation quantified associations between SE and outcomes. Multivariable linear regression tested association between poor SE and health-related quality of life controlling for significant covariates (age, diagnosis, comorbidities, worsening Functional Status Scale). Following ABI, median SE was 91.7 (interquartile range = 83.3, 95.5). Nineteen (19%) children had poor SE (< 80%). SE correlated significantly with quality of life (Spearman's correlation = .307) and Family Impact Module (Spearman's correlation = .309; both P < .01). When controlling for covariates, poor SE significantly increased risk for lower health-related quality of life (β-coefficient = -7.0; 95% confidence interval= -13.4, -0.6). One in five infants and young children with ABI have poor SE that is associated with poorer child and family health outcomes. Our study underscores the potential importance of sleep following ABI to optimize recovery and the need for additional investigation of SE in infants and young children. Klapp JM, Hall TA, Riley AR, Janzen D, Williams CN. Post-PICU sleep efficiency and quality of life in infants and toddlers with acquired brain injury. J Clin Sleep Med. 2024;20(1):75-83.

Using IT to Improve Outcomes for Children Living With Cancer (SyMon-SAYS): Protocol for a Single-Institution Waitlist Randomized Controlled Trial.

Children and adolescents with cancer may experience multiple disease- and treatment-related symptoms that negatively affect health-related quality of life. Routine symptom surveillance thus constitutes an important component of supportive care in pediatric oncology. The Symptom Monitoring and Systematic Assessment and Reporting System in Young Survivors (SyMon-SAYS) system will administer, score, interpret, and display the results of symptom assessments captured weekly using patient-reported outcomes presented via the electronic health record (EHR) portal between clinic visits in oncology ambulatory settings, when patients are likely to be more symptomatic. This study is testing a digital system for routine symptom surveillance that includes EHR-based reports to clinicians and alerts for severe symptoms. In this randomized trial, we are examining the effects of the SyMon-SAYS system on perceived barriers to symptom management, self-efficacy, and symptom severity. Better self-management and timely clinical intervention to address symptoms promote adherence to treatment plans, strengthen child and parent self-efficacy, improve interactions between children, parents, and their clinical providers, and optimize clinical outcomes. The SyMon-SAYS system is integrated into the EHR to streamline the presentation of symptom scores and delivery of alerts for severe symptoms to clinicians using EHR (Epic) messaging functionalities. Children (aged 8 to 17 years) complete the weekly symptom assessment and review the symptom report by logging into the patient portal (Epic MyChart). This single-institution waitlist randomized controlled trial is recruiting 200 children (aged 8-17 years) with cancer and their parents, guardians, or caregivers. Participating dyads are randomly assigned to receive the intervention over 16 weeks (Group A: 16-week SyMon-SAYS intervention; Group B: 8-week usual care and then an 8-week SyMon-SAYS intervention). Analyses will (1) evaluate the efficacy of SyMon-SAYS at week 8 and the maintenance of those effects at week 16; (2) evaluate factors associated with those efficacy outcomes, including contextual factors, adherence to the SyMon-SAYS intervention, demographic characteristics, and clinical factors; and (3) evaluate predictors of adherence to the SyMon-SAYS intervention and preference of SyMon-SAYS versus usual care. Data collection is currently in progress. We hypothesize that at 8 weeks, those receiving the SyMon-SAYS intervention will report decreased parent-perceived barriers to managing their children's symptoms, increased parent and child self-efficacy, decreased child symptom burden, and ultimately better child health-related quality of life, compared to waitlist controls. Feasibility, acceptability, and engagement from the perspectives of the children with cancer, their parents, and their clinicians will be examined using mixed methods. We anticipate that this system will facilitate prompt identification of problematic symptoms. Additionally, we hypothesize that with the availability of graphical symptom reports over time, and timely provider responses, children or parents will become better informed and take an active role in managing their symptoms, which will further improve clinical outcomes. ClinicalTrials.gov NCT04789720; https://clinicaltrials.gov/study/NCT04789720. DERR1-10.2196/50993.

Relationships Between Parental Anxiety and Child Quality of Life in Advanced Childhood Cancer.

Background: Family factors, such as household income and parental psychosocial distress, have been associated with quality of life in children with cancer. However, relationships between parent anxiety and child health-related quality of life (HRQoL) have not been evaluated in children with advanced cancer. Objective: To examine relationships between parent anxiety and both parent-reported and child self-reported HRQoL for children with advanced cancer. Method: Children (aged 5-17 years) with relapsed or refractory cancer and their parents participated in this single-institution cross-sectional study. Parents completed measures of their own baseline anxiety (State-Trait Anxiety Inventory-Trait [STAI-T] form) and their ill child's HRQoL (PedsQL Generic and PedsQL Cancer, parent report). Children completed age-specific PedsQL Generic and PedsQL Cancer, child report. Spearman's rho coefficients assessed correlations between total parent STAI-T score and both parent-reported and child-reported HRQoL scales. Results: Twenty children (Mage = 9.5 years, 50% female) and their 20 parents (90% mothers) participated. The strongest and statistically significant (p < .05) correlations were inverse associations between parental trait anxiety and parent-reported child psychosocial HRQoL (rs = -.54), emotional functioning (rs = -.49), school functioning (rs = -.45), and child pain and hurt (rs = -.45). Correlations of parental anxiety with all dimensions of child-reported HRQoL were generally smaller (rs < .40), positive, and not statistically significant (p > .05). Discussion: Given the inverse correlations between parental anxiety and child psychosocial HRQoL, assessment of parent mental health needs and access to interventions should be provided routinely for parents of children with advanced cancer.

User-perceived impact of long-term mechanical assisted cough in paediatric neurodisability.

To (1) compare the perceived benefit of long-term mechanical insufflation-exsufflation (MI-E) of children with neuromuscular disorders (NMDs) and central nervous system (CNS) disorders, including health care needs and treatment routines and (2) describe the children's health-related quality of life (HRQoL). This cross-sectional study used a questionnaire and memory card data to assess the perceived benefit of MI-E via the Visual Analogue Scale (VAS; 10 maximum), willingness to pause treatment, level of health care needs before and after MI-E initiation, and the children's treatment routines. A DISABKIDS questionnaire assessed HRQoL (100 maximum). Seventy-three children using MI-E participated (42 males, median [interquartile range {IQR}] age 10 years 2 months [6 years 3 months-14 years 1 month]), 47 with NMDs (such as spinal muscular atrophy and Duchenne muscular dystrophy) and 26 with CNS disorders (such as cerebral palsy, encephalitis, neurometabolic and other diseases). The median (IQR) VAS score for the perceived benefit of MI-E therapy at stable state and respiratory tract infection were 9 (6-10) and 10 (8.5-10) respectively. Sixty-two per cent were reluctant or unwilling to pause MI-E therapy, with no NMD versus CNS disorder group difference. After MI-E initiation, fewer physician consultations and hospitalizations were reported by the group with NMDs. The MI-E routine was similar in both groups. The mean (SD) HRQoL score for 26 of 51 eligible children was 71 (16.7). MI-E treatment was generally perceived as beneficial and performed equally in both diagnostic groups. HRQoL was in line with children with a moderate-to-severe chronic condition. Mechanical insufflation-exsufflation (MI-E) was generally perceived as beneficial by the children and parents. The reported benefit of MI-E was higher among daily than sporadic MI-E users. The MI-E treatment routine did not differ between diagnostic groups. The health-related quality of life in this neuropaediatric population was in line with that of children with other moderate-to-severe chronic conditions.

Evaluation of RuralkidsGPS; A Novel Integrated Paediatric Care Coordination Model of Care in Rural Australia - a Mixed-Methods Study Protocol.

The Kids Guided Personalised Service (KidsGPS) is an integrated model of care coordination for children and young people (CYP) living with medical complexity. After successful implementation in an urban setting, the model of care will be rolled-out at scale to four rural regions in New South Wales, Australia to establish RuralKidsGPS. This paper describes the approach and methods for the outcome and implementation evaluation of RuralKidsGPS. The evaluation aims to assess health, economic and implementation outcomes and processes whilst identifying barriers and enablers to inform future rollouts. Measures of health service utilisation (primary outcome), child health related quality of life and parent/carer experiences will be assessed. The implementation evaluation will occur alongside the outcomes evaluation and is underpinned by the Consolidated Framework for Implementation Research and informed by validated quantitative measures and qualitative interviews with patients, families, healthcare providers and service managers. An economic analysis will determine incremental cost effectiveness ratios for the new model of care using health service utilisation data. RuralKidsGPS, if effective, has the potential to improve equity of access to integrated care for CYP and their families and this protocol may inform other evaluations of similar models of care delivered at scale.

Association between children's health-related quality of life, healthcare costs and socioeconomic position: Results from a longitudinal Australian-based study

BackgroundEquity in healthcare service access and use are national goals and principal indicators of health system performance. Whilst it is important to understand how healthcare costs, socioeconomic position (SEP), and children's well-being are associated, limited literature explores how these covary. AimsTo explore the associations between families' SEP and (1) children's health-related quality of life (HRQoL) from 4 to 13 years; and (2) healthcare costs from 0 to 13 years; as well as (3) contributing factors that influence children's HRQoL and healthcare costs. MethodsData were drawn from the Longitudinal Study of Australian Children (LSAC). We used the parent-reported Pediatric Quality of Life Inventory 4.0 to measure children's HRQoL from 4 to 13 years. Healthcare costs to government from 0 to 13 years were measured using the government administrative data (Medicare) which includes records of both medical and pharmaceutical service utilisation. We used a composite of income, parental education, and occupation to measure SEP. We used linear and mixed effects modelling to explore associations between families' SEP and children's HRQoL or government healthcare costs, including the effects of predictors for these associations. Analyses were weighted and accounted for the survey design. ResultsCompared to children from high SEP families, those from low SEP families had lower HRQoL (from age 6–13 years, p = 0.001) and their families incurred higher government healthcare costs (from birth to 13 years). This association was confirmed using the mixed effects model (p = 0.054). Child special healthcare needs, being a single-parent or having parental stress were related to poorer children's HRQoL and higher government healthcare costs. Living in regional and remote areas was related to lower government healthcare costs. ConclusionUp to 13 years of age, children with low SEP used more health services but had lower HRQoL than those from high SEP families. These findings highlight a need to support children from low SEP families to improve their health and wellbeing.

Health-related quality of life of children with Williams syndrome and caregivers in China.

Williams syndrome (WS) is a rare genetic disorder that impacts multiple systems and may cause developmental delays. These medical and developmental issues impose a heavy burden on affected children and their families. However, there was no study on children's health-related quality of life (HRQoL) with WS and only two studies about family quality of life globally. Therefore, the primary purpose of this study was to assess the HRQoL of children with WS and their caregivers in China, and the secondary purpose was to identify the potential determinants of children's and caregivers' HRQoL. In total, 101 children and caregivers were included. We applied the proxy-reported PedsQL 4.0 Generic Core Module (PedsQL GCM) and PedsQL 3.0 Family Impact Module (FIM) to measure the HRQoL of children and caregivers. Additionally, we collected information on a comprehensive set of social demographic and clinical characteristics. Differences in HRQoL scores across subgroups were assessed by two-independent-samples t-tests, one-way ANOVA, and post hoc tests. We also calculated effect sizes to indicate clinical relevance. Multivariate linear regression models were applied to assess the potential determinants of HRQoL. We found that the HRQoL of children with WS and their caregivers was dramatically worse than the norm average scores of the healthy controls of children published in previous studies. Paternal educational level, household income, and the perceived financial burden significantly influenced the HRQoL of both children and families (p-values < 0.05). Multivariate linear regression analysis showed that the perceived financial burden was independently associated with family quality of life (p-values < 0.05)., and the presence of sleeping problem was independently associated with children's HRQoL (p-value = 0.01). We call for attention from policymakers and other stakeholders on the health status and well-being of children with WS and their families. Supports are needed to relieve psychosocial distress and financial burden.