Implementing neurodevelopmental follow-up care for children with congenital heart disease: A scoping review with evidence mapping.

AIM: To identify and map evidence describing components of neurodevelopmental follow-up care for children with congenital heart disease (CHD). METHOD: This was a scoping review of studies reporting components of neurodevelopmental follow-up programmes/pathways for children with CHD. Eligible publications were identified through database searches, citation tracking, and expert recommendations. Two independent reviewers screened studies and extracted data. An evidence matrix was developed to visualize common characteristics of care pathways. Qualitative content analysis identified implementation barriers and enablers. RESULTS: The review included 33 studies. Twenty-one described individual care pathways across the USA (n = 14), Canada (n = 4), Australia (n = 2), and France (n = 1). The remainder reported surveys of clinical practice across multiple geographical regions. While heterogeneity in care existed across studies, common attributes included enrolment of children at high-risk of neurodevelopmental delay; centralized clinics in children's hospitals; referral before discharge; periodic follow-up at fixed ages; standardized developmental assessment; and involvement of multidisciplinary teams. Implementation barriers included service cost/resourcing, patient burden, and lack of knowledge/awareness. Multi-level stakeholder engagement and integration with other services were key drivers of success. INTERPRETATION: Defining components of effective neurodevelopmental follow-up programmes and care pathways, along with enhancing and expanding guideline-based care across regions and into new contexts, should continue to be priorities. WHAT THIS PAPER ADDS: Twenty-two different neurodevelopmental follow-up care pathways/programmes were published, originating from four countries. Twelve additional publications described broad practices for neurodevelopmental follow-up across regions Common attributes across eligibility, service structure, assessment processes, and care providers were noted. Studies reported programme acceptability, uptake, cost, and effectiveness. Implementation barriers included service cost/resourcing, patient burden, and lack of knowledge/awareness.

Mapping Kansas City cardiomyopathy, Seattle Angina, and minnesota living with heart failure to the MacNew-7D in patients with heart disease.

INTRODUCTION: The Kansas City Cardiomyopathy Questionnaire (KCCQ), Seattle Angina Questionnaire (SAQ), and Minnesota Living with Heart Failure Questionnaire (MLHFQ) are widely used non-preference-based instruments that measure health-related quality of life (QOL) in people with heart disease. However, currently it is not possible to estimate quality-adjusted life-years (QALYs) for economic evaluation using these instruments as the summary scores produced are not preference-based. The MacNew-7D is a heart disease-specific preference-based instrument. This study provides different mapping algorithms for allocating utility scores to KCCQ, MLHFQ, and SAQ from MacNew-7D to calculate QALYs for economic evaluations. METHODS: The study included 493 participants with heart failure or angina who completed the KCCQ, MLHFQ, SAQ, and MacNew-7D questionnaires. Regression techniques, namely, Gamma Generalized Linear Model (GLM), Bayesian GLM, Linear regression with stepwise selection and Random Forest were used to develop direct mapping algorithms. Cross-validation was employed due to the absence of an external validation dataset. The study followed the Mapping onto Preference-based measures reporting Standards checklist. RESULTS: The best models to predict MacNew-7D utility scores were determined using KCCQ, MLHFQ, and SAQ item and domain scores. Random Forest performed well for item scores for all questionnaires and domain score for KCCQ, while Bayesian GLM and Linear Regression were best for MLHFQ and SAQ domain scores. However, models tended to over-predict severe health states. CONCLUSION: The three cardiac-specific non-preference-based QOL instruments can be mapped onto MacNew-7D utilities with good predictive accuracy using both direct response mapping techniques. The reported mapping algorithms may facilitate estimation of health utility for economic evaluations that have used these QOL instruments.

Navigating the challenges of imposter participants in online qualitative research: lessons learned from a paediatric health services study.

BACKGROUND: The growth in online qualitative research and data collection provides several advantages for health service researchers and participants, including convenience and extended geographic reach. However, these online processes can also present unexpected challenges, including instances of participant fraud or scam behaviour. This study describes an incident of participant fraud identified during online focus group discussions and interviews for a PhD health services research project on paediatric neurodevelopmental care. METHODS: We aimed to recruit carers of Australian children with neurodevelopmental disorders. Potential participants were recruited via a publicly available social media advert on Facebook offering $50 AUD compensation. Those who expressed interest via email (n = 254) were sent a pre-interview Qualtrics survey to complete. We identified imposters at an early stage via inconsistencies in their self-reported geographical location and that captured by the survey as well as recognition of suspicious actions before, during and after focus group discussions and interviews. RESULTS: Interest in participation was unexpectedly high. We determined that all potential participants were likely imposters, posing as multiple individuals and using different IP addresses across Nigeria, Australia, and the United States. In doing so, we were able to characterise several "red flags" for identifying imposter participants, particularly those posing as multiple individuals. These comprise a combination of factors including large volumes and strange timings of email responses, unlikely demographic characteristics, short or vague interviews, a preference for nonvisual participation, fixation on monetary compensation, and inconsistencies in reported geographical location. Additionally, we propose several strategies to combat this issue such as providing proof of location or eligibility during recruitment and data collection, examining email and consent form patterns, and comparing demographic data with regional statistics. CONCLUSIONS: The emergent risk of imposter participants is an important consideration for those seeking to conduct health services research using qualitative approaches in online environments. Methodological design choices intended to improve equity and access for the target population may have an unintended consequence of improving access for fraudulent actors unless appropriate risk mitigation strategies are also employed. Lessons learned from this experience are likely to be valuable for novice health service researchers involved in online focus group discussions and interviews.

Cost-effectiveness of left atrial appendage closure for stroke prevention in atrial fibrillation: a systematic review appraising the methodological quality.

BACKGROUND: The increasing global prevalence of atrial fibrillation (AF) has led to a growing demand for stroke prevention strategies, resulting in higher healthcare costs. High-quality economic evaluations of stroke prevention strategies can play a crucial role in maximising efficient allocation of resources. In this systematic review, we assessed the methodological quality of such economic evaluations. METHODS: We searched electronic databases of PubMed, EMBASE, CINAHL, Cochrane Central Register of Controlled Trials, and Econ Lit to identify model-based economic evaluations comparing the left atrial appendage closure procedure (LAAC) and oral anticoagulants published in English since 2000. Data on study characteristics, model-based details, and analyses were collected. The methodological quality was evaluated using the modified Economic Evaluations Bias (ECOBIAS) checklist. For each of the 22 biases listed in this checklist, studies were categorised into one of four groups: low risk, partial risk, high risk due to inadequate reporting, or high risk. To gauge the overall quality of each study, we computed a composite score by assigning + 2, 0, - 1 and - 2 to each risk category, respectively. RESULTS: In our analysis of 12 studies, majority adopted a healthcare provider or payer perspective and employed Markov Models with the number of health states varying from 6 to 16. Cost-effectiveness results varied across studies. LAAC displayed a probability exceeding 50% of being the cost-effective option in six out of nine evaluations compared to warfarin, six out of eight evaluations when compared to dabigatran, in three out of five evaluations against apixaban, and in two out of three studies compared to rivaroxaban. The methodological quality scores for individual studies ranged from 10 to - 12 out of a possible 24. Most high-risk ratings were due to inadequate reporting, which was prevalent across various biases, including those related to data identification, baseline data, treatment effects, and data incorporation. Cost measurement omission bias and inefficient comparator bias were also common. CONCLUSIONS: While most studies concluded LAAC to be the cost-effective strategy for stroke prevention in AF, shortcomings in methodological quality raise concerns about reliability and validity of results. Future evaluations, free of these shortcomings, can yield stronger policy evidence.

Preferences for Neurodevelopmental Follow-Up Care for Children: A Discrete Choice Experiment.

INTRODUCTION: Identifying and addressing neurodevelopmental delays in children can be challenging for families and the healthcare system. Delays in accessing services and early interventions are common. The design and delivery of these services, and associated outcomes for children, may be improved if service provision aligns with families' needs and preferences for receiving care. The aim of this study is to identify families' preferences for neurodevelopmental follow-up care for children using an established methodology. METHODS: We used a discrete choice experiment (DCE) to elicit families' preferences. We collected data from families and caregivers of children with neurodevelopmental needs. The DCE process included four stages. In stage 1, we identified attributes and levels to be included in the DCE using literature review, interviews, and expert advice. The finalised attributes were location, mode of follow-up, out-of-pocket cost per visit, mental health counselling for parents, receiving educational information, managing appointments, and waiting time. In stage 2, we generated choice tasks that contained two alternatives and a 'neither' option for respondents to choose from, using a Bayesian d-efficient design. These choice tasks were compiled in a survey that also included demographic questions. We conducted pre- and pilot tests to ensure the functionality of the survey and obtain priors. In stage 3, the DCE survey was administered online. We received 301 responses. In stage 4, the analysis was conducted using a latent class model. Additionally, we estimated the relative importance of attributes and performed a scenario analysis. RESULTS: Two latent classes were observed. More families with full-time employees, higher incomes, postgraduate degrees, and those living in metropolitan areas were in class 1 compared with class 2. Class 1 families preferred accessing local public health clinics, face-to-face follow-up, paying AUD100 to AUD500, mental health support, group educational activities, health service-initiated appointments, and waiting < 3 months. Class 2 families disliked city hospitals when compared with private, preferred paying AUD100 or no cost, and had similar preferences regarding mental health support and wait times as class 1. However, no significant differences were noted in follow-up modality, receiving educational information, and appointment management. The relative importance estimation suggested that location was most important for class 1 (28%), whereas for class 2, cost accounted for nearly half of the importance when selecting an alternative. The expected uptake of follow-up care, estimated under three different hypothetical scenarios, may increase by approximately 24% for class 2 if an 'ideal' scenario taking into account preferences was implemented. CONCLUSION: This study offers insights into aspects that may be prioritised by health services and policymakers to improve the design and delivery of neurodevelopmental follow-up care for children. The findings may enhance the organisation and functioning of existing care programmes; and therefore, improve the long-term outcomes of children with neurodevelopmental needs and their families.

Stakeholders' preferences for the design and delivery of virtual care services: A systematic review of discrete choice experiments.

This systematic review aimed to synthesise evidence from discrete choice experiments (DCEs) eliciting preferences for virtual models of care, as well as to assess the quality of those DCEs and compare the relative preferences for different stakeholder groups. Articles were included if published between January 2010 and December 2022. Data were synthesised narratively, and attributes were assessed for frequency, significance, and relative importance using a semi-quantitative approach. Overall, 21 studies were included encompassing a wide range of virtual care modalities, with the most common setting being virtual consultations for outpatient management of chronic conditions. A total of 135 attributes were identified and thematically classified into six categories: service delivery, service quality, technical aspects, monetary aspects, health provider characteristics and health consumer characteristics. Attributes related to service delivery were most frequently reported but less highly ranked. Service costs were consistently significant across all studies where they appeared, indicating their importance to the respondents. All studies examining health providers' preferences reported either system performance or professional endorsement attributes to be the most important. Substantial heterogeneity in attribute selection and preference outcomes were observed across studies reporting on health consumers' preferences, suggesting that the consideration of local context is important in the design and delivery of person-centred virtual care services. In general, the experimental design and analysis methods of included studies were clearly reported and justified. An improvement was observed in the quality of DCE design and analysis in recent years, particularly in the attribute development process. Given the continued growth in the use of DCEs within healthcare settings, further research is needed to develop a standardised approach for quantitatively synthesising DCE findings. There is also a need for further research on preferences for virtual care in post-pandemic contexts, where emerging evidence suggests that preferences may differ to those observed in pre-pandemic times.

Preferences in the Design and Delivery of Neurodevelopmental Follow-Up Care for Children: A Systematic Review of Discrete Choice Experiments.

Neurodevelopmental disorders are a significant cause of morbidity. Early detection of neurodevelopmental delay is essential for timely diagnosis and intervention, and it is therefore important to understand the preferences of parents and clinicians for engaging with neurodevelopmental surveillance and follow-up care. Discrete choice experiment (DCE) may be an appropriate method for quantifying these preferences. This review systematically examined how DCEs have been designed and delivered in studies examining neurodevelopmental care of children and identified the preferred attributes that have been reported. PubMed, Embase, CINAHL, and Scopus databases were systematically searched. Studies were included if they used DCE to elicit preferences for a neurodevelopmental follow-up program for children. Two independent reviewers conducted the title and abstract and full-text screening. Risk of bias was assessed using a DCE-specific checklist. Findings were presented using a narrative synthesis. A total of 6618 records were identified and 16 papers were included. Orthogonal (n=5) and efficient (n=5) experimental designs were common. There was inconsistent reporting of design-related features. Analysis was primarily completed using mixed logit (n=6) or multinomial logit (n=3) models. Several key attributes for neurodevelopmental follow-up care were identified including social, behavioral and emotional support, therapy, waiting time, and out-of-pocket costs. DCE has been successfully used as a preference elicitation method for neurodevelopmental-related care. There is scope for improvement in the design and analysis of DCE in this field. Nonetheless, attributes identified in these studies are likely to be important considerations in the design and implementation of programs for neurodevelopmental care.

Long-term Patient and Health Service Outcomes of Ablation and Antiarrhythmic Drugs in Atrial Fibrillation: A Comparative Systematic Review.

Atrial fibrillation (AF) is a prevalent problem worldwide and a common cause of hospitalization, poor quality of life, and increased mortality. Although several treatments are used, the use of ablation and antiarrhythmic drug therapy has increased in the past decade. However, debate continues on the most suitable option for heart rhythm control in patients. Previous studies have largely focused on short-term outcome effects of these treatments. This systematic review aims to determine the effect of ablation compared to antiarrhythmic drugs for AF on long-term patient and health service outcomes of mortality, hospitalization, and quality of life. Three databases were systematically searched-studies were included if they reported long-term outcomes of more than 12 months comparing the 2 treatments. Title and abstract screening and subsequent full-text screening was done by 2 reviewers. Data were extracted from the final studies identified. The details of the search were recorded according to Preferred Reporting Items for Systematic Reviews and Meta-Analyses report. A total of 2224 records were identified. After removing duplicates and screening the titles and abstracts, 68 records required full-text screening. Finally, 12 papers were included in the analysis. Eight studies reported mortality indicating ablation was superior, 2 studies reported hospitalization with opposing outcomes, and 5 quality of life studies indicating ablation was a better treatment. In studies assessing long-term outcomes, beyond 12 months, following ablation or rhythm control drugs for AF, most found a lower risk of death and greater improvement in quality of life in the ablation group.