对罕见疾病患者的协调护理:CONCORD混合方法研究

Stephen Morris, E. Hudson, Lara Bloom, L. Chitty, N. Fulop, A. Hunter, Jennifer Jones, J. Kai, L. Kerecuk, M. Kokocinska, K. Leeson-Beevers, P. L. Ng, Sharon Parkes, A. Ramsay, Amy Simpson, A. Sutcliffe, Christine Taylor, H. Walton
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引用次数: 2

摘要

如果在普通人群中影响不到1/2000人,则被定义为罕见。有限的证据表明,对受罕见疾病影响的人的护理协调不力。调查英国是否以及如何协调对罕见病患者的护理,以及受罕见病影响的人希望如何协调护理。一项混合方法研究,包括(1)范围界定审查,以制定协调护理的定义并确定协调护理的组成部分(n = 154项研究);(2) 一项探索性的定性访谈研究,以了解缺乏协调护理的影响(n = 15名参与者);(3) 一项针对受罕见护理协调经验影响人群的全国性调查(n = 1457名参与者);(4) 协调偏好的离散选择实验(n = 996名参与者);(5) 罕见病协同护理分类法的发展(n = 79名参与者);以及(6)对提供协调护理的成本进行审查。为受罕见疾病影响的人提供医疗服务,包括提供社会护理和第三部门护理。患有罕见疾病的成年患者、患有罕见疾病儿童或成人的父母/看护人以及参与照顾罕见疾病患者的医疗保健专业人员(如医生、护士和专职医疗保健专业人士)。英国对包括的罕见疾病或人们居住的地方没有限制。参与者是从患者和提供者网络和组织中抽取的。制定了罕见疾病协调护理的定义。对罕见病患者的护理被发现没有得到很好的协调。例如,760名患有罕见病的成年患者中,只有12%的人报告说他们有正式的护理协调员,32%的人报告他们去了专科中心,10%的人报告称他们有护理计划。患者、父母/护理人员和医疗保健专业人员都希望更好地协调护理,患者和父母/护理者报告的偏好与医疗保健专业人士报告的偏好存在一些差异。我们对罕见疾病的护理协调分类概述了六个领域:(1)组织护理的方式,(2)组织团队的方式。不可能捕捉到每一种罕见疾病患者的经历。如果研究参与者与受罕见疾病影响的人群存在系统性差异,我们在研究中的抽样策略可能存在偏见。成本分析有限。有证据表明,对罕见病患者缺乏协调一致的护理。这可能会对患者和家人的身心健康以及他们的经济状况产生负面影响。使用本研究中开发的分类法,进一步的研究将有利于开发可行、临床有效和成本效益高的护理协调模型。本研究注册为NIHR临床研究网络投资组合参考号41132,研究注册参考号researchregistry6351和综合研究应用系统参考号254400。该项目由国家卫生研究所(NIHR)卫生和社会护理提供研究计划资助,并将在《卫生和社会保健提供研究》上全文发表;第10卷第5期。有关更多项目信息,请访问NIHR期刊图书馆网站。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Co-ordinated care for people affected by rare diseases: the CONCORD mixed-methods study
A condition is defined as rare if it affects fewer than 1 in 2000 people in the general population. Limited evidence suggests that care is poorly co-ordinated for people affected by rare conditions. To investigate if and how care of people with rare conditions is co-ordinated in the UK, and how people affected by rare conditions would like care to be co-ordinated. A mixed-methods study comprising (1) a scoping review to develop a definition of co-ordinated care and identify components of co-ordinated care (n = 154 studies); (2) an exploratory qualitative interview study to understand the impact of a lack of co-ordinated care (n = 15 participants); (3) a national survey among people affected by rare conditions of experiences of care co-ordination (n = 1457 participants); (4) a discrete choice experiment of preferences for co-ordination (n = 996 participants); (5) the development of a taxonomy of co-ordinated care for rare conditions (n = 79 participants); and (6) a review of costs of providing co-ordinated care. Health services for people affected by rare conditions, including gatekeeping to social care provision and third-sector care. Adult patients with rare conditions, parents/carers of children or adults with rare conditions and health-care professionals (e.g. doctors, nurses and allied health professionals) involved in the care of people with rare conditions. No limits were set on the rare conditions included or where people live in the UK. Participants were sampled from patient and provider networks and organisations. A definition of co-ordinated care for rare conditions was developed. Care for people affected by rare diseases was found to be not well co-ordinated. For example, only 12% of 760 adult patients affected by a rare disease reported that they had a formal care co-ordinator, 32% reported that they attended a specialist centre and 10% reported that they had a care plan. Patients, parents/carers and health-care professionals all would like care to be better co-ordinated, with some differences in preferences reported by patients and parents/carers and those reported by health-care professionals. Our taxonomy of care co-ordination for rare conditions outlined six domains: (1) ways of organising care, (2) ways of organising teams, (3) responsibilities, (4) how often care appointments and co-ordination take place, (5) access to records and (6) mode of communication. It was not possible to capture the experiences of people affected by every rare condition. Our sampling strategy in the study may have been biased if study participants were systematically different from the population affected by rare conditions. The cost analysis was limited. There is evidence of a lack of co-ordinated care for people affected by rare diseases. This can have a negative impact on the physical and mental health of patients and families, and their financial well-being. Further research would be beneficial to develop feasible, clinically effective and cost-effective models of care co-ordination, using the taxonomy developed in this study. This study is registered as NIHR Clinical Research Network Portfolio reference number 41132, Research Registry reference number research registry6351 and Integrated Research Application System reference number 254400. This project was funded by the National Institute for Health Research (NIHR) Health and Social Care Delivery Research programme and will be published in full in Health and Social Care Delivery Research; Vol. 10, No. 5. See the NIHR Journals Library website for further project information.
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