Title: Effective healthcare delivery in rare rheumatic disease: evaluating models of care for systemic vasculitis
Funder: Versus Arthritis

Abstract: Background Effective organisation and delivery of healthcare services for individuals with rare autoimmune rheumatic disease is essential to ensure timely access to specialist expertise and optimise clinical outcomes. Yet, patients report feeling lost in the system, struggling to navigate through a healthcare system not designed to manage complex disease. For example, individuals with systemic vasculitis, a group of devastating but treatable multi-organ illnesses, frequently experience diagnostic delays, uncoordinated care and inconsistent access to relevant expertise. Our preliminary studies indicate clinically important differences in management practices and outcomes even among specialised centres. Numerous models of care for systemic vasculitis exist but it remains unclear what works best, for whom, and in what circumstances. There is increasing momentum within the UK to implement commitments made in the UK Rare Disease Strategy, and within Europe to improve services for patients with rare diseases. However, these efforts have largely been focused on genetic conditions. To date, no studies have characterised the organisation of care for those with rare rheumatic disease and how this relates to outcomes for all patients at a national level. Aims and objectives This innovative study seeks to address this key evidence gap. We aim to identify key elements underpinning effective care to inform development of effective, equitable vasculitis services and support for patients. Study design The proposal consists of three work packages; Work package 1. Geographical analysis of clinical outcomes and patterns of healthcare use in systemic vasculitis This will build on a pilot study examining co-morbidity in ANCA-associated vasculitis (AAV) in select specialist services using linked datasets in Scotland. Focusing on AAV and Giant Cell Arteritis (GCA), we will identify all patients in Scotland with the relevant International Classification of Diseases (ICD)-10 codes alongside age, sex and postcode matched controls. Patient records will be linked to national administrative databases and geographical variation in outcomes e.g. mortality, cancer, co-morbidities, infections and use of healthcare resources quantified. Work package 2. Organisation, delivery and experiences of care An online survey of vasculitis services across the UK will characterise a number of features of each service including size, structure, human resources, facilities, processes of care and management across a range of different healthcare contexts. For services in Scotland, survey data will be compared with outcome data obtained in Work package 1 to explore associations between care organisation and health outcomes. We will also conduct six in-depth case studies across the UK to understand patient and staff experiences of care. Using a whole systems approach we will undertake a multi-level analysis at macros, meso and micro levels. Observation and semi-structured interview data will be gathered from a diverse range of stakeholders. Novel patient ethnography methods will be used in three cases to gain first hand insights into the patient journey. Interviews with patients will be used to produce a new section on systemic vasculitis on the public website healthtalk.org, a freely accessible online resource for patients and their families. Work package 3. Recommendations to inform development of a new service delivery model We will bring key stakeholders (patients, healthcare professionals, organisations and commissioners) in two co-design workshops. Guided by experience based co-design principles we will present our findings from Work packages 1 and 2, explore what better care looks like and co-produce recommendations to inform policy and practice. At the end of this project we will have; 1) Explored geographical variation in outcome for patients with systemic vasculitis at national level in Scotland 2) Mapped current vasculitis service provision and processes of care across the UK 3) Explored the relationship between health outcomes and care delivery 4) Conducted an in-depth analysis of the patient journey, focusing on experiences of care and areas of unmet need. Key deliverables 1) Set of recommendations to directly inform national and international reconfiguration of vasculitis services 2) Healthtalk online website on systemic vasculitis for patients and their families This proposal addresses a key strategic aim of ARUK and is especially timely given increasing momentum within the UK and Europe to optimise services for those with rare diseases. Results from this study will be immediately relevant to inform national and international service development of vasculitis services. Using systemic vasculitis as an exemplar of a rare, adult onset rheumatic disease and the shared challenges of delivering care to those with complex multi-system diseases, we anticipate findings will be applicable to other rare rheumatic diseases. Furthermore, the multi-disciplinary expertise and methodological approach used in this study will act as a blueprint that can be upscaled and applied to more common rheumatic conditions to enhance the organisation, equality and effectiveness of care for those living with rheumatic disease.


Research activity codes: 7.1 INDIVIDUAL CARE NEEDS;7.3 MANAGEMENT AND DECISION MAKING
Health categories: INFLAMMATORY AND IMMUNE SYSTEM