Share and protect our health data: an evidence based approach to rare disease patients’ perspectives on data sharing and data protection - quantitative survey and recommendations

Abstract

Background The needs and benefits of sharing health data to advance scientific research and improve clinical benefits have been well documented in recent years, specifically in the field of rare diseases where knowledge and expertise are limited and patient populations are geographically dispersed. Understanding what patients want and need from rare disease research and data sharing is important to ensure their participation and engagement in the process, and to ensure that these wishes and needs are embedded within research design. EURORDIS-Rare Diseases Europe regularly surveys the rare disease community to identify its perspectives and needs on a number of issues in order to represent rare disease patients and be their voice within European and International initiatives and policy developments. Here, we present key findings from a large quantitative survey conducted with patients with rare diseases and family members as part of a continuous evidence-based advocacy process developed at EURORDIS. The aim of this survey was to explore patient and family perspectives on data sharing and data protection in research and healthcare settings and develop relevant recommendations to support shaping of future data sharing initiatives in rare disease research. This survey, translated into 23 languages, was carried out via the Rare Barometer Programme and was designed to be accessible to a diverse population with a wide range of education backgrounds. It was widely disseminated via patient organisations worldwide to ensure that a wide range of voices and experiences were represented. Main findings Rare disease patients, regardless of the severity of their disease and their socio-demographic profile, are clearly supportive of data sharing to foster research and improve healthcare. However, rare disease patients’ willingness to share their data does come with specific requirements in order to respect their privacy, choices and needs for information regarding the use of their data. Conclusions To ensure sustainability and success of international data sharing initiatives in health and research for rare diseases, appropriate legislations need to be implemented and multi-stakeholder efforts need to be pursued to foster cultural and technological changes enabling the systematic integration of patients’ preferences regarding sharing of their own health data. Electronic supplementary material The online version of this article (10.1186/s13023-019-1123-4) contains supplementary material, which is available to authorized users.

Sharing of Clinical Trial Data and Samples: The Cancer Patient Perspective

“…Fifth, patients in this study expressed only few concerns about the for-profit/not-for-profit nature of organizations, explaining that even if pharmaceutical companies are driven by profit, their profit is made by developing products that benefit patients, thus ultimately all medical research serves the same purpose. This finding is somewhat contrary to the results from previously published quantitative studies, which indicated that research participants and rare diseases patients were more likely or comfortable to allow their data to be shared with not-for-profit stakeholders (e.g., academic researchers, health care professionals, non-profit and patient organizations) than with researchers in for-profit companies or insurance companies (31,32,35,36). Yet, most participants in our study did mention that they deplore a lack of collaboration and sharing between researchers because of commercial reasons.…”

“…Fourth, all but two patients expressed their trust in ethics committees taking up the task of intermediary decision maker. In a previous quantitative study with 2,005 patients with rare diseases, about half of the respondents indicated that they would allow an ethics committee to decide on their behalf (35). Our finding reflects the practices as prescribed by ethical recommendations such as the Helsinki Declaration, although not echoed in all legal frameworks since the EU data protection framework does not stipulate any intermediary form of control for secondary re-use of sensitive data.…”

A systematic literature review of health consumer attitudes towards secondary use and sharing of health administrative and clinical trial data: a focus on privacy, trust, and transparency

“…Other studies focused on health consumers' attitudes to secondary data usage in individuals: attending US Veterans Affairs (VA) facilities [40] or recently discharged from tertiary care [15], or with arthritis and other chronic conditions [36]. Others were in the setting of human immunodeficiency virus (HIV) [49], breast cancer (BC), colon cancer (CC) [35], or heterogeneous cancers [19,20], acquired immune deficiency syndrome (AIDS), or multiple sclerosis (MS), or mental health concerns [23], presenting with rare diseases [16,17], in adults or parents of children with cystic fibrosis (CF), sickle cell disease (SCD), or diabetes mellitus (DM) [35], or in adults with potentially stigmatising health conditions (DM, hypertension, chronic depression, alcoholism, HIV, BC, or lung cancer) [46].…”

“…If a personal connection with the research team is established, then it is easier for individuals to form a trusting relationship [23]. The highest levels of trust was placed in the doctor [16,26,46], the National Health Service (NHS) [26], and hospitals [16,46], while the lowest trust was in commercial organisations [26], pharmaceutical companies and insurance companies [46], or for-profit organisations [16]. An individual's trust in an organisation was a determinant of what level of control they preferred over their data [40] and their willingness to participate in research [42], with trust overcoming concerns about privacy and confidentiality [49].…”

A scoping review and proposed workflow for multi-omic rare disease research

“…Furthermore, there is a need for discussion on ensuring the data we generate is publicly available, whilst protecting patient confidentiality, to enable large scale collaborative efforts, a phenomenon which would be particularly helpful for diagnosing currently undiagnosed patients. Such a discussion and development of resources should involve continuous consultation with patients and their family members [16,153].…”