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.

Autor:	Hollick RJ; Epidemiology Group, Aberdeen Centre for Arthritis and Musculoskeletal Health, University of Aberdeen, Aberdeen, UK. Electronic address: rhollick@abdn.ac.uk., James WRG; Epidemiology Group, Aberdeen Centre for Arthritis and Musculoskeletal Health, University of Aberdeen, Aberdeen, UK., Nicoll A; Health Services Research Unit, University of Aberdeen, Aberdeen, UK., Locock L; Health Services Research Unit, University of Aberdeen, Aberdeen, UK., Black C; Aberdeen Centre for Health Data Science, University of Aberdeen, Aberdeen, UK; NHS Grampian, Foresterhill, Aberdeen, UK., Dhaun N; British Heart Foundation/University Centre for Cardiovascular Science, University of Edinburgh, Edinburgh, UK., Egan AC; Trinity Health Kidney Centre, Tallaght University Hospital, Dublin, Ireland., Fluck N; NHS Grampian, Foresterhill, Aberdeen, UK., Laidlaw L; Epidemiology Group, Aberdeen Centre for Arthritis and Musculoskeletal Health, University of Aberdeen, Aberdeen, UK., Lanyon PC; Department of Rheumatology, Nottingham University Hospitals NHS Trust, Nottingham, UK; Lifespan and Population Health, School of Medicine, University of Nottingham, Nottingham, UK., Little MA; Trinity Kidney Centre, Trinity Translational Medicine Institute, Trinity College Dublin, Dublin, Ireland., Luqmani RA; Nuffield Department of Orthopaedics, Rheumatology and Musculoskeletal Science, University of Oxford, Oxford, UK., Moir L; Epidemiology Group, Aberdeen Centre for Arthritis and Musculoskeletal Health, University of Aberdeen, Aberdeen, UK., McBain M; Epidemiology Group, Aberdeen Centre for Arthritis and Musculoskeletal Health, University of Aberdeen, Aberdeen, UK., Basu N; School of Infection and Immunity, University of Glasgow, Glasgow, UK.
Jazyk:	angličtina
Zdroj:	The Lancet. Rheumatology [Lancet Rheumatol] 2024 Jun; Vol. 6 (6), pp. e361-e373.
DOI:	10.1016/S2665-9913(24)00082-1
Abstrakt:	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. Competing Interests: Declaration of interest NB has received speaker fees and research funding from VSL Vifor. RJH has received funding for the present study from Versus Arthritis; is clinical lead of the Scottish Systemic Vasculitis Managed Clinical Network; and has received speaker fees from CSL Vifor. PCL is co-chair of the Rare Autoimmune Rheumatic Disease Alliance (RAIRDA), the joint national clinical lead for the Rheumatology, Getting It Right First Time programme, NHS England, and the clinical lead for Rare Diseases, National Disease Registration Service, NHS England; has received speaker fees from CSL Vifor and Pfizer; and expenses for attending conferences and research funding from CSL Vifor. MAL has received speaker fees from CSL Vifor. LL's research time for this study was supported by the Health Services Research Unit, University of Aberdeen, which received core funding from the Chief Scientist's Office (Scotland). All other authors declare no competing interests. (Copyright © 2024 The Author(s). Published by Elsevier Ltd. This is an Open Access article under the CC BY 4.0 license.)
Databáze:	MEDLINE
Externí odkaz:	Zobrazit plný text záznamu