Identifying key health system components associated with improved outcomes to inform the re-configuration of services for adults with rare autoimmune rheumatic diseases: a mixed-methods study.

BACKGROUND: Adults with rare autoimmune rheumatic diseases face unique challenges and struggles to navigate health-care systems designed to manage common conditions. Evidence to inform an optimal service framework for their care is scarce. Using systemic vasculitis as an exemplar, we aimed to identify and explain the key service components underpinning effective care for rare diseases. METHODS: In this mixed-methods study, data were collected as part of a survey of vasculitis service providers across the UK and Ireland, interviews with patients, and from organisational case studies to identify key service components that enable good care. The association between these components and patient outcomes (eg, serious infections, mortality) and provider outcomes (eg, emergency hospital admissions) were examined in a population-based data linkage study using routine health-care data obtained from patients with antineutrophil cytoplasmic antibody (ANCA)-associated vasculitis from national health datasets in Scotland. We did univariable and multivariable analyses using Bayesian poisson and negative binomial regression to estimate incident rate ratios (IRRs), and Cox proportional hazards models to estimate hazard ratios (HRs). People with lived experiences were involved in the research and writing process. FINDINGS: Good care was characterised by service components that supported timely access to services, integrated care, and expertise. In 1420 patients with ANCA-associated vasculitis identified from national health datasets, service-reported average waiting times for new patients of less than 1 week were associated with fewer serious infections (IRR 0·70 [95% credibility interval 0·55-0·88]) and fewer emergency hospital admissions (0·78 [0·68-0·92]). Nurse-led advice lines were associated with fewer serious infections (0·76 [0·58-0·93]) and fewer emergency hospital admissions (0·85 [0·74-0·96]). Average waiting times for new patients of less than 1 week were also associated with reduced mortality (HR 0·59 [95% credibility interval 0·37-0·93]). Cohorted clinics, nurse-led clinics, and specialist vasculitis multi-disciplinary team meetings were associated with fewer serious infections (IRR 0·75 [0·59-0·96] for cohorted clinics; 0·65 [0·39-0·84] for nurse-led clinics; 0·72 [0·57-0·90] for specialist vasculitis multi-disciplinary team meetings) and emergency hospital admissions (0·81 [0·71-0·91]; 0·75 [0·65-0·94]; 0·86 [0·75-0·96]). Key components were characterised by their ability to overcome professional tensions between specialties. INTERPRETATION: Key service components associated with important health outcomes and underpinning factors were identified to inform initiatives to improve the design, delivery, and effectiveness of health-care models for rare autoimmune rheumatic diseases. FUNDING: Versus Arthritis.

The role of framing, agency and uncertainty in a focus-divide dilemma.

How to prioritise multiple objectives is a common dilemma of daily life. A simple and effective decision rule is to focus resources when the tasks are difficult, and divide when tasks are easy. Nonetheless, in experimental paradigms of this dilemma, participants make highly variable and suboptimal strategic decisions when asked to allocate resources to two competing goals that vary in difficulty. We developed a new version in which participants had to choose where to park a fire truck between houses of varying distances apart. Unlike in the previous versions of the dilemma, participants approached the optimal strategy in this task. Three key differences between the fire truck version and previous versions of the task were investigated: (1) Framing (whether the objectives are familiar or abstract), by comparing a group who placed cartoon trucks between houses to a group performing the same task with abstract shapes; (2) Agency (how much of the task is under the participants' direct control), by comparing groups who controlled the movement of the truck to those who did not; (3) Uncertainty, by adding variability to the driving speed of the truck to make success or failure on a given trial more difficult to predict. Framing and agency did not influence strategic decisions. When adding variability to outcomes, however, decisions shifted away from optimal. The results suggest choices become more variable when the outcome is less certain, consistent with exploration of response alternatives triggered by an inability to predict success.

Inefficient Eye Movements: Gamification Improves Task Execution, But Not Fixation Strategy.

Decisions about where to fixate are highly variable and often inefficient. In the current study, we investigated whether such decisions would improve with increased motivation. Participants had to detect a discrimination target, which would appear in one of two boxes, but only after they chose a location to fixate. The distance between boxes determines which location to fixate to maximise the probability of being able to see the target: participants should fixate between the two boxes when they are close together, and on one of the two boxes when they are far apart. We "gamified" this task, giving participants easy-to-track rewards that were contingent on discrimination accuracy. Their decisions and performance were compared to previous results that were gathered in the absence of this additional motivation. We used a Bayesian beta regression model to estimate the size of the effect and associated variance. The results demonstrate that discrimination accuracy does indeed improve in the presence of performance-related rewards. However, there was no difference in eye movement strategy between the two groups, suggesting this improvement in accuracy was not due to the participants making more optimal eye movement decisions. Instead, the motivation encouraged participants to expend more effort on other aspects of the task, such as paying more attention to the boxes and making fewer response errors.