Collaborative Research and Development of a Novel, Patient-Centered Digital Platform (MyEyeSite) for Rare Inherited Retinal Disease Data: Acceptability and Feasibility Study.
Collaborative Research and Development of a Novel, Patient-Centered Digital Platform (MyEyeSite) for Rare Inherited Retinal Disease Data: Acceptability and Feasibility Study.
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DOI：
10.2196/21341
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发表时间：
2022-01-31
影响因子：
2.2
通讯作者：
Cammack J
中科院分区：
文献类型：
--
作者：
Gilbert RM;Sumodhee D;Pontikos N;Hollyhead C;Patrick A;Scarles S;Van Der Smissen S;Young RM;Nettleton N;Webster AR;Cammack J
遗传性视网膜疾病（IRD）是英国和其他国家儿童和工作年龄成年人失明的主要原因，具有显着的社会经济影响。然而，根据定义，IRD 数据很少，因此，该患者群体的研究服务不足。研究人员需要大量的稀有数据才能在该领域取得进展，例如通过基因疗法的开发。面临的挑战是如何以最有效的方式找到这些数据并将其提供给研究人员。 MyEyeSite 是一项研究合作项目，旨在为罕见 IRD 患者设计和开发一个数字平台（MyEyeSite 平台），使患者、医生和研究人员能够汇总和共享专业眼部健康数据。该平台的一个重要组成部分是 MyEyeSite 患者应用程序，它将为 IRD 患者提供与系统交互的方式，特别是整理、管理和共享他们的个人专业 IRD 数据，以用于研究和自己的医疗保健。本研究旨在通过一项以患者为中心的协作研究来测试 MyEyeSite 平台在目标 IRD 人群中的可接受性和可行性。定性数据通过焦点小组和研讨会产生，定量数据通过对 IRD 患者的调查获得。参与者是通过莫菲尔德眼科医院国家卫生服务 (NHS) 基金会信托基金和国家健康研究所 (NIHR) 莫菲尔德生物医学研究中心的诊所通过患者和公众参与数据库招募的。我们的 IRD 焦点小组样本 (n=50) 强调了以下主题： 对英国 NHS 内当前数据共享系统的不满；由于增加了对这些专业数据的访问，对 MyEyeSite 患者应用程序的潜在好处抱有积极的期望；以及对数据安全的担忧，包括在 NHS 之外可能不道德地使用数据。在接受调查的 80 名参与者中，68 名 (85%) 人有动力在眼部护理中发挥更积极的作用，并使用安全技术（例如网络应用程序或移动应用程序）共享他们的数据以用于研究目的。这项研究表明，IRD 患者非常积极地积极参与管理自己的研究数据和自己的眼睛护理。它展示了让 IRD 患者参与 MyEyeSite 平台示例详细设计的可行性，IRD 研讨会患者的意见在确定设计和原型的功能性和可访问性方面发挥着关键作用。针对罕见健康数据问题开发以用户为中心的技术解决方案不仅有可能使 IRD 社区患者受益，而且有可能使其他罕见疾病患者受益。
Inherited retinal diseases (IRDs) are a leading cause of blindness in children and working age adults in the United Kingdom and other countries, with an appreciable socioeconomic impact. However, by definition, IRD data are individually rare, and as a result, this patient group has been underserved by research. Researchers need larger amounts of these rare data to make progress in this field, for example, through the development of gene therapies. The challenge has been how to find and make these data available to researchers in the most productive way. MyEyeSite is a research collaboration aiming to design and develop a digital platform (the MyEyeSite platform) for people with rare IRDs that will enable patients, doctors, and researchers to aggregate and share specialist eye health data. A crucial component of this platform is the MyEyeSite patient application, which will provide the means for patients with IRD to interact with the system and, in particular, to collate, manage, and share their personal specialist IRD data both for research and their own health care. This study aims to test the acceptability and feasibility of the MyEyeSite platform in the target IRD population through a collaborative patient-centered study. Qualitative data were generated through focus groups and workshops, and quantitative data were obtained through a survey of patients with IRD. Participants were recruited through clinics at Moorfields Eye Hospital National Health Service (NHS) Foundation Trust and the National Institute for Health Research (NIHR) Moorfields Biomedical Research Centre through their patient and public involvement databases. Our IRD focus group sample (n=50) highlighted the following themes: frustration with the current system regarding data sharing within the United Kingdom’s NHS; positive expectations of the potential benefits of the MyEyeSite patient application, resulting from increased access to this specialized data; and concerns regarding data security, including potentially unethical use of the data outside the NHS. Of the surveyed 80 participants, 68 (85%) were motivated to have a more active role in their eye care and share their data for research purposes using a secure technology, such as a web application or mobile app. This study demonstrates that patients with IRD are highly motivated to be actively involved in managing their own data for research and their own eye care. It demonstrates the feasibility of involving patients with IRD in the detailed design of the MyEyeSite platform exemplar, with input from the patient with IRD workshops playing a key role in determining both the functionality and accessibility of the designs and prototypes. The development of a user-centered technological solution to the problem of rare health data has the potential to benefit not only the patient with IRD community but also others with rare diseases.