Is the Comparator in Your Diagnostic Cost-Effectiveness Model "Standard of Care"? Recommendations from Literature Reviews and Expert Interviews on How to Identify and Operationalize It.

OBJECTIVES: This research aimed to develop best-practice recommendations for identifying the "standard of care" (SoC) and integrate it when it is the comparator in diagnostic economic models (SoC comparator). METHODS: A multi-methods approach comprising 2 pragmatic literature reviews and 9 expert interviews was used. Experts rated their agreement with draft recommendations based on the authors' analysis of the reviews. These were refined iteratively to produce final recommendations. RESULTS: Fourteen best-practice recommendations are provided. Care pathway mapping (using quantitative, qualitative, or mixed-methods approaches) should be used for identifying the SoC comparator. Guidelines analysis can be integrated with expert opinion to identify pathway variability and discrepancies from clinical practice. For integrating the SoC comparator into the model, recommendations around structure, input sourcing, data aggregation and reporting, input uncertainty, and model variability are presented. For example, modelers should consider that the reference standard is not synonymous with the SoC, and the SoC may not be the only comparator. The comparator limitations should be discussed with clinical experts, but elicitation of its diagnostic accuracy is not recommended. Probabilistic sensitivity analysis is recommended when evaluating the overall input uncertainty, and deterministic sensitivity analysis is useful when there is high model uncertainty or SoC variability. Consensus could not be reached for some topics (eg, the role of real-world data, model averaging, and alternative model structures), but the reported discussions provide points for consideration. CONCLUSIONS: To our knowledge, this is the first guidance to support modelers when identifying and operationalizing the SoC comparator in diagnostic cost-effectiveness models.

A novel, nurse-led 'one stop' clinic for patients with liver cirrhosis results in fewer liver-related unplanned readmissions and improved survival.

OBJECTIVE: Delivering effective secondary preventive and integrated care has the potential to break the revolving-door phenomenon of frequent readmissions in patients with advanced chronic liver disease. To address this, we launched the Care Coordination of Liver Disease (CCoLD) pilot, a novel nurse-led cirrhosis clinic in Western Sydney. METHODS AND ANALYSIS: Following an index presentation to Blacktown or Mount Druitt hospitals (BMDH), patients (n = 89, matched by age, sex, and MELD-NA) were consecutively either followed up by the CCoLD clinical nurse consultant (intervention cohort) or received standard care (control cohort). Controlled evaluation of the impact of the nurse-led clinic was carried out for a 3-month period including readmission rates, survival, and cost effectiveness. RESULTS: The inaugural nurse-led clinic led to improvement in patient-level outcomes including a reduction in unplanned liver-related readmissions (2.08% for intervention cohort vs 12.2% for control cohort, p < 0.01), and mortality at 30 days (0% for intervention cohort vs 7.3% for control cohort, p = 0.03). Similar trends were observed at 90 days from index discharge. No deaths were observed in the intervention cohort as compared to the control cohort at 90 days (0% versus 7.3%, p = 0.03), while unplanned liver-related readmissions were 10.41% for the intervention cohort vs 19.5% for the control cohort (p = 0.115). Moreover, time to readmission was significantly longer in the intervention cohort, resulting in an overall cost-effective intervention. CONCLUSION: These findings highlight the significant impact of optimised care-coordination. A nurse-led clinic can deliver patient-centred, goal-directed, and cost-effective secondary prevention and care. A multicentre randomised trial for wider evaluation of these findings is warranted.

Transitional care programs for older adults moving from hospital to home in Canada: A systematic review of text and opinion.

BACKGROUND: Investing in transitional care programs is critical for ensuring continuity of health and coordinated care for older adults transitioning across health settings. However, literature delineating the scope of transitional care programs across Canada is limited. The aim of this systematic review of text and opinion is to characterize Canadian transitional care programs for older adults transitioning from hospital to home. METHODS: Following JBI guidelines for systematic review of text and opinion, we conducted a search of Canadian grey literature sources published online between 2016 to 2023. A 3-phase search was undertaken for: 1) Canadian databases and organizational websites; 2) advanced Google search of national sources and news media reports; and 3) advanced Google search of provincial/territorial sources. Two reviewers independently screened sources for eligibility against inclusion criteria. Data were extracted by one reviewer and verified by a second. Textual data were extracted from multiple sources to characterize each transitional care program. RESULTS: Grey literature search produced a total of 17,092 text and opinion sources, identifying 119 transitional care programs in Canada. Model of care was a key characteristic defining the design and delivery of transitional care programs within community (n = 42), hospital (n = 45), and facility-based (n = 32) settings. Programs were characterized by goal, population and eligibility, setting and length of program, intervention and services, and healthcare team members. Patient, caregiver, and health system outcomes were reported for 18 programs. The province of Ontario has the most transitional care programs (n = 84) and reported outcomes, followed by British Columbia (n = 10). CONCLUSIONS: Characterizing transitional care programs is important for informing health services planning and scaling up of transitional care program models across Canada. Recognizing transitional care programs as a core health service is critical to meet the health care needs of older adults at the right time and place. TRIAL REGISTRATION: PROSPERO ID 298821.

A machine learning approach to identify important variables for distinguishing between fallers and non-fallers in older women.

Falls are a significant ongoing public health concern for older adults. At present, few studies have concurrently explored the influence of multiple measures when seeking to determine which variables are most predictive of fall risks. As such, this cross-sectional study aimed to identify those functional variables (i.e. balance, gait and clinical measures) and physical characteristics (i.e. strength and body composition) that could best distinguish between older female fallers and non-fallers, using a machine learning approach. Overall, 60 community-dwelling older women (≥65 years), retrospectively classified as fallers (n = 21) or non-fallers (n = 39), attended three data collection sessions. Data (281 variables) collected from tests in five separate domains (balance, gait, clinical measures, strength and body composition) were analysed using random forest (RF) and leave-one-variable-out partial least squares correlation analysis (LOVO PLSCA) to assess variable importance. The strongest discriminators from each domain were then aggregated into a multi-domain dataset, and RF, LOVO PLSCA, and logistic regression models were constructed to identify the important variables in distinguishing between fallers and non-fallers. These models were used to classify participants as either fallers or non-fallers, with their performance evaluated using receiver operating characteristic (ROC) analysis. The study found that it is possible to classify fallers and non-fallers with a high degree of accuracy (e.g. logistic regression: sensitivity = 90%; specificity = 87%; AUC = 0.92; leave-one-out cross-validation accuracy = 63%) using a combination of 18 variables from four domains, with the gait and strength domains being particularly informative for screening programmes aimed at assessing falls risk.

Care pathway analysis and evidence gaps in adult-onset Still's disease: interviews with experts from the UK, France, Italy, and Germany.

Introduction: Adult-onset Still's disease (AOSD) is a rare systemic inflammatory disease of unknown etiology. Published AOSD data are limited, and clinical guidelines were lacking until recently. Managing AOSD remains largely empirical with uncertainties and high variability about the optimal care pathway. Therefore, we used a qualitative approach to collect clinical judgments from the UK, Italy, France and Germany to inform the development of an agreed care pathway. Our work aimed to decrease the uncertainty associated with clinical practice, inform future research in AOSD, and help identify standardized definitions and outcomes in this population. Methods: Semi-structured interviews and thematic analysis were conducted. Eleven clinicians were interviewed between May and July 2022: four were based in Italy, three in the UK, two in France, and two in Germany. Results: In this work, we identified the structure of the typical care pathway for AOSD patients, which can be used to inform future economic models in AOSD. The general structure of the pathway was similar across countries. Non-steroidal anti-inflammatory drugs are prescribed during the diagnostic workup while an additive approach is commonly used in confirmed cases: corticosteroids, conventional synthetic disease-modifying antirheumatic drugs, then biologic disease-modifying antirheumatic drugs (bDMARDs) (dose increased before switching). For severe presentations, more aggressive approaches with higher doses and early use of bDMARDs are used. The main elements of variation among countries and clinicians were the criteria used for diagnosis; order of bDMARDs and preferential treatments for articular and systemic patients; and tests for patient monitoring. There is also a lack of standardized outcome measures making comparisons and evidence synthesis challenging. Conclusion: We identified important evidence gaps for clinical practice, e.g., reliable tests or scores predictive of disease progression and treatment outcome, and recommendations for research, e.g., reporting of compliance rates and use of the Yamaguchi criteria for clinical study inclusion. Consensus is needed around the use of the Systemic score in clinical practice and the clinical utility of this score. A standardized definition of remission is also required in AOSD, and further research should look to identify and validate the specific laboratory markers to be considered when assessing remission.

Transitional care programs in Canada for older adults transitioning from hospital to home: protocol for a systematic review of text and opinion.

OBJECTIVE: The objective of this systematic review is to identify what transitional care programs exist across Canada, including the characteristics and outcomes of these programs. INTRODUCTION: There is growing evidence of the benefits of transitional care programs to support older adults moving from hospital to home. However, there is limited literature identifying the types of transitional care programs that exist internationally and little evidence available within Canada. INCLUSION CRITERIA: Sources of gray literature published from 2016 that focus on older adults receiving services from transitional care programs to move from hospital to home in Canada will be considered for inclusion. Sources of gray literature will be excluded if interventions are targeted at adults younger than 65 years, Indigenous adults younger than 55 years, or if the primary discharge destination is not an independent community dwelling. Interventions designed for older adults waiting in hospital for long-term care placement will also be excluded from this review. METHODS: An initial limited search of Canadian national gray literature resources will be undertaken, followed by an advanced Google search of Canadian resources and news media reports. Lastly, an advanced search of Google for all 10 provinces and 3 territories will be undertaken to target examples of local transitional care programs that may not be found through a national search, such as local pilot projects, health region-specific programs, and provincial organizations. All identified sources will be retrieved and full text review of selected citations assessed in detail by 2 independent reviewers. Data about the characteristics and outcomes of transitional care programs and results will be extracted and synthesized, with a meta-aggregation approach for grading according to JBI ConQual method. SYSTEMATIC REVIEW REGISTRATION NUMBER: PROSPERO CRD42022298821.

The burden of mitochondrial disease with associated seizures: systematic literature reviews of health-related quality of life, utilities, costs and healthcare resource use data.

BACKGROUND: Mitochondrial disease is a degenerative, progressive, heterogeneous group of genetic disorders affecting children and adults. Mitochondrial disease is associated with morbidity and mortality, with predominantly neurological and neuromuscular symptoms including dystonia, weakness, encephalopathy, developmental delay and seizures. Seizures are one of the most common and severe manifestations of mitochondrial disease. These seizures are typically refractory to common anti-seizure therapies. There are no approved disease-modifying treatments for mitochondrial disease. Our objective was to conduct two systematic literature reviews to identify health-related quality of life (HRQoL), utilities, costs and healthcare resource use data in mitochondrial disease with associated seizures. METHODS: A range of databases and information sources were searched up to July 2022 to identify eligible studies. Search strategies included a range of variant terms for mitochondrial disease and HRQoL, utilities, cost and healthcare resource use outcomes. Two reviewers independently assessed articles against the eligibility criteria; studies were extracted by one reviewer and checked by a second. Risk of bias was assessed for studies reporting HRQoL data. Results were narratively assessed. RESULTS: Seven studies were eligible for the HRQoL and utilities review. The studies used different tools to report data, and despite the variability in methods, HRQoL scores across the studies showed moderate/severe disease in patients with mitochondrial disease with associated seizures. Parents of patients with mitochondrial disease with associated seizures were characterised by high total parenting stress. No studies reported utilities data. Two case reports and one retrospective review of medical records of children who died in hospital were eligible for the costs and resource use review. These provided limited information on the duration of hospital stay, in an intensive care unit (ICU), on mechanical ventilation. No studies reported costs data. CONCLUSION: These reviews highlight the limited HRQoL, utilities, costs and resource use data and the variability of instruments used in mitochondrial disease with associated seizures. However, the data available indicate that mitochondrial disease with associated seizures affects patients' and caregivers' HRQoL alike. No robust conclusion can be drawn on the impact of mitochondrial disease with associated seizures on hospital or ICU length of stay. Trial registration PROSPERO: CRD42022345005.

Burden of adult-onset Still's disease: A systematic review of health-related quality of life, utilities, costs and resource use.

Adult-onset Still's disease (AOSD) poses a not well estimated burden on patients and healthcare systems. To assess this burden, a systematic review (SR) was undertaken to identify health-related quality of life (HRQoL), utilities, costs and healthcare resource use data. Searches of twelve databases, four conferences, and three key technology assessment and regulatory agency websites were conducted in August 2022. Reference lists of retrieved SRs published since 2017 were also checked. Overall, 16 studies were eligible for inclusion. Eight studies reported HRQoL outcomes, one of which also reported utilities data. Two studies reported direct costs outcomes, and seven reported healthcare resource use data. No indirect costs were identified. A range of outcomes were reported, thus limiting the comparability of results across studies. SF-36 data were impaired in AOSD on most scales, especially those concerning physical activity. Mean SF-36 data were lower across all subscales in patients with active AOSD compared with inactive AOSD. Biologic therapy showed improvements in the SF-36 physical health summary. Utility scores (one study) were significantly lower for AOSD than for healthy controls. Limited direct economic costs data were identified but were substantial where reported. Hospital length of stay ranged from 6.1 to 23.5 days. The SR showed there is a paucity of research reporting the HRQoL and cost burden of AOSD.

Biodiversity conservation cannot afford COVID-19 communication bungles.

With COVID-19 (coronavirus disease 2019) dominating headlines, highlighting links between the pandemic and biodiversity may increase public awareness of the biodiversity crisis. However, ill-considered messages that frame nature as the problem rather than the solution could inadvertently propagate problematic narratives and undermine motivations and individual self-efficacy to conserve nature.