Abstract
Background: Adults with rare autoimmune rheumatic diseases (RAIRDs) face unique challenges and struggles to navigate healthcare systems designed to manage common conditions. Evidence to inform an optimal service framework for their care is lacking. Using systemic vasculitis as an exemplar, we aimed to identify and explain the key service components underpinning effective rare
disease care.
Methods: This mixed-methods study drew on a survey of vasculitis service providers across the UK and Ireland, interviews with patients and organisational case studies to identify key service components that enable good care. The relationship between these components and patient (e.g., serious infections, mortality), and provider (e.g., emergency hospital admissions) outcomes were
examined in a population-based data linkage study using routine healthcare data from n=1,420 patients with vasculitis in Scotland. Bayesian Poisson/Negative Binomial regression and Cox Proportional hazards models reported results as Incident Rate Ratios (IRR) or Hazard Ratios (HR), respectively. 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. Average new patient waiting times <1 week and nurse41 advice lines were associated with fewer serious infections (IRR 0·70 (0·55, 0·88); 0·76 (0·58,0·93)) and emergency hospital admissions (IRR 0·78 (0·68, 0·92); 0·85 (0·74,0·96)). Average new patient waiting times <1 week were also associated with reduced mortality (HR 0·59 (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); 0·65 (0·39, 0·84); 0·72 (0·57, 0·90)) and emergency hospital admissions (IRR 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 healthcare models for RAIRDs.
disease care.
Methods: This mixed-methods study drew on a survey of vasculitis service providers across the UK and Ireland, interviews with patients and organisational case studies to identify key service components that enable good care. The relationship between these components and patient (e.g., serious infections, mortality), and provider (e.g., emergency hospital admissions) outcomes were
examined in a population-based data linkage study using routine healthcare data from n=1,420 patients with vasculitis in Scotland. Bayesian Poisson/Negative Binomial regression and Cox Proportional hazards models reported results as Incident Rate Ratios (IRR) or Hazard Ratios (HR), respectively. 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. Average new patient waiting times <1 week and nurse41 advice lines were associated with fewer serious infections (IRR 0·70 (0·55, 0·88); 0·76 (0·58,0·93)) and emergency hospital admissions (IRR 0·78 (0·68, 0·92); 0·85 (0·74,0·96)). Average new patient waiting times <1 week were also associated with reduced mortality (HR 0·59 (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); 0·65 (0·39, 0·84); 0·72 (0·57, 0·90)) and emergency hospital admissions (IRR 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 healthcare models for RAIRDs.
| Original language | English |
|---|---|
| Journal | The Lancet Rheumatology |
| Publication status | Accepted/In press - 13 Mar 2024 |
Bibliographical note
Funding for this study was provided by Versus Arthritis (grant number 22088). The authors would like to thank all the patient contributors with lived experience of systemic vasculitis who have been involved in this study. We also wish to acknowledge our collaborators, the UKIVAS, for their support in the conceptualisation and delivery of the study, including development and distribution of the vasculitis provider survey, recruitment to case study sites and for hosting workshops to discuss study findings and inform recommendations. We are also grateful to the European Reference Network for rare immune disorders (ERN RITA) for their support with development and distribution of the survey to members. We also wish to thank the eDRIS Team (Public Health Scotland) for their involvement in obtaining approvals, provisioning, and linking data and the use of the secure analytical platform within the National Safe HavenData Availability Statement
Patient interviews are archived with the Medical Sociology & Health Experiences Researcher Group, University of Oxford. They are all copyrighted to the University of Oxford and available, under licence, to qualitative researchers for secondary analysis (subject to approval and administrative costs). Access to NHS Scotland health data is governed by the NHS Scotland Public Benefit and Privacy Panel for Health and Social Care (HSC-PBPP). Access to the underlying pseudonymised healthdata used in this study is by application to the HSC-PBPP panel. All code used in the analyses, from data processing to completed results, is available for review at: https://github.com/warren630 james/VOICES_DataProcessingAndAnalysis