Paediatrics (RCH) - Research Publications
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ItemPsychometric Performance Comparison of the Adapted versus Original Versions of the EQ-5D-Y-3L and -Y-5L in Proxy Respondents for 2-to 4-Year-Olds(ADIS INT LTD, 2024-01-18)INTRODUCTION: Few preference-weighted instruments are available to measure health-related quality of life in young children (2-4 years of age). The EQ-5D-Y-3L and EQ-5D-Y-5L were recently modified for this purpose. OBJECTIVE: The aim of this study was to test the psychometric properties of these adapted versions for use with parent proxies of children aged 2-4 years and to compare their performance with the original versions. It was hypothesised that the adapted instrument wording would result in improved psychometric performance. METHODS: Survey data of children aged 2-4 years were obtained from the Australian Paediatric Multi-Instrument Comparison study. Distributional and psychometric properties tested included feasibility, convergence, distribution of level scores, ceiling effects, known-group validity (Cohen's D effect sizes for prespecified groups defined by the presence/absence of special healthcare needs [SHCNs]), test-retest reliability (intraclass correlation coefficients [ICCs]), and responsiveness (standardised response mean [SRM] effect sizes for changes in health). Level sum scores were used to provide summary outcomes. Supplementary analysis using utility scores (from the Swedish EQ-5D-Y-3L value set) were conducted for the adapted and original EQ-5D-Y-3L, and no value sets were available for the EQ-5D-Y-5L. RESULTS: A total of 842 parents of children aged 2-4 years completed the survey. All instruments were easy to complete. There was strong convergence between the adapted and original EQ-5D-Y-3L and EQ-5D-Y-5L. The adapted EQ-5D-Y-3L and adapted EQ-5D-Y-5L showed more responses in the severe levels of the five EQ-5D-Y dimensions, particularly in the usual activity and mobility dimensions (EQ-5D-Y-5L: mobility level 1: adapted n = 478 [83%], original n = 253 [94%]; mobility level 4/5: adapted n = 17 [2.9%], original n = 4 [1.5%)]). The difference in the distribution of responses was more evident in children with SHCNs. Assessment of known-group validity showed a greater effect size for the adapted EQ-5D-Y-3L and adapted EQ-5D-Y-5L compared with the original instruments (EQ-5D-Y-5L: adapted Cohen's D = 1.01, original Cohen's D = 0.83) between children with and without SHCNs. The adapted EQ-5D-Y-3L and adapted EQ-5D-Y-5L showed improved reliability at 4-week follow-up, with improved ICCs (EQ-5D-Y-5L: adapted ICC = 0.83, original ICC = 0.44). The responsiveness of all instruments moved in the hypothesised direction for better or worse health at follow-up. Probability of superiority analysis showed little/no differences between the adapted and original EQ-5D-Y-3L and EQ-5D-Y-5L. Supplementary psychometric analysis of the adapted and original EQ-5D-Y-3L using utilities showed comparable findings with analyses using level sum scores. CONCLUSIONS: The findings suggest improved psychometric performance of the adapted version of the EQ-5D-Y-3L and EQ-5D-Y-5L in children aged 2-4 years compared with the original versions.
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ItemComparative Psychometric Performance of Common Generic Paediatric Health-Related Quality of Life Instrument Descriptive Systems: Results from the Australian Paediatric Multi-Instrument Comparison Study(ADIS INT LTD, 2023-11-13)OBJECTIVE: The aim of this study was to compare the psychometric performance of common generic paediatric health-related quality-of-life instrument descriptive systems (PedsQL generic core 4.0, EQ-5D-Y-3L, EQ-5D-Y-5L, Child Health Utility 9D [CHU9D], Assessment of Quality of Life 6D [AQoL-6D], and Health Utilities Index Mark 3 [HUI3]) by child age, report type, and health status. METHODS: Data for children aged 5-18 years were from the Australian Paediatric Multi-Instrument Comparison study. Ceiling effects, test-retest reliability, known-group validity, convergent and divergent validity, and responsiveness were assessed in the total sample and by child age (5-12 years vs 13-18 years), report type (self- vs proxy report), and health status. Instruments were scored using an exploratory level sum score (LSS) approach. RESULTS: Survey data were available for 5945 children, with follow-up data available for 2346 children. The EQ-5D-Y-3L demonstrated ceiling effects. The PedsQL, EQ-5D-Y-3L, EQ-5D-Y-5L, and CHU9D demonstrated acceptable test-retest reliability. All instruments demonstrated known-group, convergent, and divergent validity. The EQ-5D-Y-3L, EQ-5D-Y-5L, and CHU9D demonstrated responsiveness to improvements in health and the PedsQL, EQ-5D-Y-3L, EQ-5D-Y-5L, and CHU9D to worsening health. The AQoL-6D and HUI3 had inconclusive test-retest reliability and responsiveness evidence due to small sample size. Importantly, ceiling effects, test-retest reliability and responsiveness varied by subgroup. CONCLUSION: Results reflect instrument performance using LSSs, which may differ to utility scores. In the total sample, the EQ-5D-Y-5L and CHU9D descriptive systems demonstrated evidence of good performance (i.e., meeting prespecified criteria) across all psychometric attributes tested. Performance varied by child age and report type, indicating room for considerations by population and study.
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ItemComparing the Psychometric Performance of Generic Paediatric Health-Related Quality of Life Instruments in Children and Adolescents with ADHD, Anxiety and/or Depression(Adis, 2024)Objective: The aim of this study was to examine the validity, reliability and responsiveness of common generic paediatric health-related quality of life (HRQoL) instruments in children and adolescents with mental health challenges. Methods: Participants were a subset of the Australian Paediatric Multi-Instrument Comparison (P-MIC) study and comprised 1013 children aged 4–18 years with attention-deficit/hyperactivity disorder (ADHD) (n = 533), or anxiety and/or depression (n = 480). Participants completed an online survey including a range of generic paediatric HRQoL instruments (PedsQL, EQ-5D-Y-3L, EQ-5D-Y-5L, CHU9D) and mental health symptom measures (SDQ, SWAN, RCADS-25). A subset of participants also completed the HUI3 and AQoL-6D. The psychometric performance of each HRQoL instrument was assessed regarding acceptability/feasibility; floor/ceiling effects; convergent validity; known-group validity; responsiveness and test–retest reliability. Results: The PedsQL, CHU9D, EQ-5D-Y-3L and EQ-5D-Y-5L showed similarly good performance for acceptability/feasibility, known-group validity and convergent validity. The CHU9D and PedsQL showed no floor or ceiling effects and fair–good test–retest reliability. Test–retest reliability was lower for the EQ-5D-Y-3L and EQ-5D-Y-5L. The EQ-5D-Y-3L showed the highest ceiling effects, but was the top performing instrument alongside the CHU9D on responsiveness to improvements in health status, followed by the PedsQL. The AQoL-6D and HUI3 showed good acceptability/feasibility, no floor or ceiling effects, and good convergent validity, yet poorer performance on known-group validity. Responsiveness and test–retest reliability were not able to be assessed for these two instruments. In subgroup analyses, performance was similar for all instruments for acceptability/feasibility, known-group and convergent validity, however, relative strengths and weaknesses for each instrument were noted for ceiling effects, responsiveness and test–retest reliability. In sensitivity analyses using utility scores, performance regarding known-group and convergent validity worsened slightly for the EQ-5D-Y-3L and CHU9D, though improved slightly for the HUI3 and AQoL-6D. Conclusions: While each instrument showed strong performance in some areas, careful consideration of the choice of instrument is advised, as this may differ dependent on the intended use of the instrument, and the age, gender and type of mental health condition of the population in which the instrument is being used. Trial Registration: ANZCTR—ACTRN12621000657820.
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ItemDistinct developmental trajectories of health-related quality of life for boys and girls throughout childhood and adolescence; a national level longitudinal study(BMC, 2023-08-01)BACKGROUND: To identify and describe distinct developmental trajectories of health-related quality of life (HRQoL) in a national level Australian population sample, overall and separately for boys and girls. METHODS: Data were from the Longitudinal Study of Australian Children (LSAC). Participants were children aged 4-5 years recruited in 2004 and followed through to age 16-17 years in 2016, and their caregivers. Group-based trajectory modelling was used to identify groups of children that follow qualitatively distinct developmental trajectories of HRQoL. RESULTS: Three distinct trajectories were identified for the total sample: (1) high-stable (52.2% of children); (2) middle-stable (38.0%); and (3) low-declining (9.8%). These trajectories differed for boys, who saw increasing HRQoL in the highest trajectory group; a middle-stable trajectory; and declining and rebounding HRQoL in the lowest trajectory group. In contrast, girls saw no increasing or rebounding trajectories; approximately half of girls had high-stable HRQoL and the remaining half had either steadily or rapidly declining HRQoL from age 4-5 to 16-17 years. CONCLUSIONS: Our results highlight the importance of considering the distinct trajectories for girls and boys and not relying on population mean levels of HRQoL for decision-making. The presence of developmentally distinct trajectories of HRQoL, and differences in the trajectories faced by boys and girls, should be considered when assessing the effectiveness of treatments and interventions impacting upon HRQoL throughout childhood and adolescence. Failure to account for these pre-existing trajectories may over- or under-estimate treatment effects.
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ItemNo Preview AvailableIdentifying and responding to family adversity in Australian community and primary health settings: a multi-site cross sectional study(Frontiers Media S.A., 2023-09-13)BACKGROUND: Unaddressed family adversity has potentially modifiable, negative biopsychosocial impacts across the life course. Little is known about how Australian health and social practitioners identify and respond to family adversity in community and primary health settings. OBJECTIVE: To describe, in two Australian community health services: (1) the number of adversities experienced by caregivers, (2) practitioner identification of caregivers experiencing adversity, (3) practitioner response to caregivers experiencing adversity, and (4) caregiver uptake of referrals. METHODS: Survey of caregivers of children aged 0-8 years attending community health services in Victoria and New South Wales (NSW). Analysis described frequencies of caregiver self-reported: (1) experiences of adversity, (2) practitioner identification of adversity, (3) practitioner response to adversity, and (4) referral uptake. Analyses were sub-grouped by three adversity domains and site. RESULTS: 349 caregivers (Victoria: n = 234; NSW: n = 115) completed the survey of whom 88% reported experiencing one or more family adversities. The median number of adversities was 4 (2-6). Only 43% of participants were directly asked about or discussed an adversity with a practitioner in the previous 6 months (Victoria: 30%; NSW: 68%). Among caregivers experiencing adversity, 30% received direct support (Victoria: 23%; NSW: 43%), and 14% received a referral (Victoria: 10%; NSW: 22%) for at least one adversity. Overall, 74% of caregivers accepted referrals when extended. CONCLUSION: The needs of Australian families experiencing high rates of adversity are not systematically identified nor responded to in community health services. This leaves significant scope for reform and enhancement of service responses to families experiencing adversity.
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ItemNo Preview AvailableWill a fee-for-service payment for a young people's health assessment in general practice increase the detection of health risk behaviours and health conditions? Protocol for a cluster randomised controlled trial (RAd Health Trial)(BMJ PUBLISHING GROUP, 2023-08)INTRODUCTION: Adolescence is a period of major transition in physical, cognitive, social and emotional development, and the peak time for the onset of mental health conditions, substance use disorders and sexual and reproductive health risks. Prevention and treatment during this time can improve health and well-being now and into the future. However, despite clinical guidelines recommending annual preventive health assessments for young people, health professionals cite lack of consultation time and adequate funding as key barriers. This trial aims to determine whether a specific fee-for-service ('rebate payment') for a young person's health assessment, is effective and cost-effective at increasing the detection and management of health risk behaviours and conditions among young people. METHODS AND ANALYSIS: This cluster randomised controlled trial will be conducted in Australian general practice. 42 general practices (clusters) will be randomly allocated 1:1 to either an intervention arm where general practitioners receive a rebate payment for each annual health assessment undertaken for 14-24-year-olds during a 2 year study period, or a control arm (no rebate). The rebate amount will be based on the Medical Benefits Schedule (Australia's list of health professional services subsidised by the Australian Government) currently available for similar age-based assessments. Our primary outcome will be the annual rate of risk behaviours and health conditions recorded in the patient electronic health record (eg, alcohol/drug use, sexual activity and mental health issues). Secondary outcomes include the annual rate of patient management activities related to health risks and conditions identified (eg, contraception prescribed, sexually transmitted infection tests ordered). A process evaluation will assess acceptability, adoption, fidelity and sustainability of the rebate; an economic evaluation will assess its cost-effectiveness. Analyses will be intention-to-treat. ETHICS AND DISSEMINATION: Ethics approval has been obtained from University of Melbourne Human and Research Ethics Committee (2022-23435-29990-3). Findings will be published in peer-reviewed journals. TRIAL REGISTRATION NUMBER: ACTRN12622000114741.
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ItemAna o 3 sIgE and diagnostic algorithms reduce cost of cashew allergy diagnosis in children compared with skin prick test: A cost comparison analysis(WILEY, 2022-08)BACKGROUND: In the absence of a clear clinical history of reaction, diagnosis of cashew allergy using skin prick tests (SPT) or cashew-specific IgE requires a high number of oral food challenges (OFC). By using Ana o 3 sIgE alone, or a two-step diagnostic algorithm using cashew sIgE followed by Ana o 3 sIgE, there is a reduced need for OFC. We aimed to perform a cost comparison for both of these approaches compared with cashew SPT alone. METHODS: Pooled individual-level data from 6 studies were used to determine diagnostic accuracy and OFC rate. Two studies used cashew SPT (n = 567, 198 allergic), with 95% positive and negative predictive values of ≥12 mm and <3 mm. Four studies were included in the pathways for Ana o 3 sIgE alone or a 2-step algorithm incorporating cashew and Ana o 3 sIgE (n = 271, 156 allergic). Cut-offs used were ≥8.5kUA/L and ≤0.1kUA/L for cashew sIgE and ≥0.35kUA/L and ≤0.1kUA/L for Ana o 3 sIgE. Costs were constructed based on unit prices from hospital inpatient admissions, expenses incurred by families, individual patient data on allergic reaction types and rates, and adrenaline autoinjector carriage, applying a health system perspective. RESULTS: Modeled data through the Ana o 3 pathway resulted in a 46.43% cost reduction (€307,406/1000 patients) compared with using cashew SPT alone (€573,854/1000 patients). The 2-step algorithm resulted in a 44.94% cost reduction compared with SPT alone (€315,952.82/1000 patients). Both the Ana o 3 pathway and 2-step algorithm resulted in a 79%-80% reduction in OFCs compared with SPT. CONCLUSIONS: Using Ana o 3 as a standalone test for cashew allergy diagnosis or a 2-step algorithm incorporating cashew sIgE and Ana o 3 sIgE is accurate and results in a large reduction in both OFCs and health system costs compared with cashew SPT alone.
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ItemOut-of-hospital health care costs of childhood food allergy in Australia: A population-based longitudinal study(WILEY, 2022-11)BACKGROUND: Australia has one of the highest prevalence of childhood food allergy in the world, but there are no data on its economic burden in Australia. METHODS: We used data from the HealthNuts study, a population-based longitudinal study undertaken in Melbourne, Australia. Infants were recruited at age 12 months between Sept 2007 and Aug 2011 with food allergy diagnosed using oral food challenges. Health care costs of out-of-hospital services were collected through data linkage to Australia's universal health insurance scheme Medicare. Two-part model was used to compare costs after controlling for potential confounders. RESULTS: 2919 children were included, and 390 (13.4%) had challenge-confirmed food allergy at age 1 year. Compared with children without food allergy, children with food allergy had significantly higher costs for GP visits, specialist visits, tests, and prescriptions in the first four years of life. The total Medicare cost associated with food allergy from age 1 to 4 years was estimated to be AUD$889.7 (95% CI $566.1-$1188.3) or €411.0 (95% CI €261.5-€549.0) per child. This was projected into an annual Medicare cost of AUD$26.1 million (95% CI $20.1-$32.3 million) or €12.1 (95% CI €9.3-€14.9 million) based on population size in 2020. CONCLUSIONS: Childhood food allergy causes considerable Medicare costs for out-of-hospital services in the first four years after birth in Australia. These findings can help anticipate the financial impact on the health care system associated with childhood food allergy, act as a useful costing resource for future evaluations, and inform management of childhood food allergy internationally.
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ItemAre We Agreed? Self- Versus Proxy-Reporting of Paediatric Health-Related Quality of Life (HRQoL) Using Generic Preference-Based Measures: A Systematic Review and Meta-Analysis(ADIS INT LTD, 2022-11)OBJECTIVE: The aim of this study was to examine the level of agreement between self- and proxy-reporting of health-related quality of life (HRQoL) in children (under 18 years of age) using generic preference-based measures. METHODS: A systematic review of primary studies that reported agreement statistics for self and proxy assessments of overall and/or dimension-level paediatric HRQoL using generic preference-based measures was conducted. Where available, data on intraclass correlation coefficients (ICCs) were extracted to summarise overall agreement levels, and Cohen's kappa was used to describe agreement across domains. A meta-analysis was also performed to synthesise studies and estimate the level of agreement between self- and proxy-reported paediatric overall and domain-level HRQoL. RESULTS: Of the 30 studies included, 25 reported inter-rater agreement for overall utilities, while 17 reported domain-specific agreement. Seven generic preference-based measures were identified as having been applied: Health Utilities Index (HUI) Mark 2 and 3, EQ-5D measures, Child Health Utility 9 Dimensions (CHU9D), and the Quality of Well-Being (QWB) scale. A total of 45 dyad samples were included, with a total pooled sample of 3084 children and 3300 proxies. Most of the identified studies reported a poor inter-rater agreement for the overall HRQoL using ICCs. In contrast to more observable HRQoL domains relating to physical health and functioning, the inter-rater agreement was low for psychosocial-related domains, e.g., 'emotion' and 'cognition' attributes of both HUI2 and HUI3, and 'feeling worried, sad, or unhappy' and 'having pain or discomfort' domains of the EQ-5D. Parents demonstrated a higher level of agreement with children relative to health professionals. Child self- and proxy-reports of HRQoL showed lower agreement in cancer-related studies than in non-cancer-related studies. The overall ICC from the meta-analysis was estimated to be 0.49 (95% confidence interval 0.34-0.61) with poor inter-rater agreement. CONCLUSION: This study provides evidence from a systematic review of studies reporting dyad assessments to demonstrate the discrepancies in inter-rater agreement between child and proxy reporting of overall and domain-level paediatric HRQoL using generic preference-based measures. Further research to drive the inclusion of children in self-reporting their own HRQoL wherever possible and limiting the reliance on proxy reporting of children's HRQoL is warranted.
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ItemThe Australian and New Zealand Fontan Registry Quality of Life Study: Protocol for a population-based assessment of quality of life among people with a Fontan circulation, their parents, and siblings(BMJ PUBLISHING GROUP, 2022-09)INTRODUCTION: Advances in the care of patients with single-ventricle congenital heart disease have led to a new generation of individuals living with a Fontan circulation. For people with Fontan physiology, physical, psychological and neurodevelopmental challenges are common. The objective of this study is to describe and develop a deeper understanding of the factors that contribute to quality of life (QOL) among children, adolescents and adults living with a Fontan circulation across Australia and New Zealand, their parents and siblings. METHODS AND ANALYSIS: This article presents the protocol for the Australian and New Zealand Fontan Registry (ANZFR) QOL Study, a cross-sectional, population-based study designed to examine QOL among people of all ages with a Fontan circulation, their parents and siblings. Study eligibility criteria includes (1) individuals with a Fontan circulation aged ≥6 years, at least 12 months post-Fontan procedure and enrolled in the ANZFR; (2) parents of individuals enrolled in the ANZFR; and (3) siblings aged ≥6 years of an individual enrolled in the ANZFR. A novel, online research platform is used to distribute personalised assessments tailored to participant age and developmental stage. A suite of validated psychometric self-report and parent-proxy report instruments capture potential correlates and predictors of QOL, including symptoms of psychological distress, personality attributes, coping and cognitive appraisals, family functioning, healthcare experiences and costs, access to emotional support and socioeconomic factors. Clinical characteristics are captured via self-report and parent-proxy report, as well as the ANZFR. Descriptive analyses and multilevel models will be used to examine QOL across groups and to investigate potential explanatory variables. ETHICS AND DISSEMINATION: Approval has been obtained from all relevant Human Research Ethics Committees (HRECs), including the Sydney Children's Hospitals Network and the Royal Children's Hospital Melbourne HRECs. Study findings will be published in peer-reviewed journals and presented at national and international meetings and seminars.