Patients' Willingness to Provide Their Clinical Data for Research Purposes and Acceptance of Different Consent Models: Findings From a Representative Survey of Patients With Cancer.

BACKGROUND: Secondary use of clinical data for biomedical research purposes holds great potential for various types of noninterventional, data-driven studies. Patients' willingness to support research with their clinical data is a crucial prerequisite for research progress. OBJECTIVE: The aim of the study was to learn about patients' attitudes and expectations regarding secondary use of their clinical data. In a next step, our results can inform the development of an appropriate governance framework for secondary use of clinical data for research purposes. METHODS: A questionnaire was developed to assess the willingness of patients with cancer to provide their clinical data for biomedical research purposes, considering different conditions of data sharing and consent models. The Cancer Registry of the German federal state of Baden-Württemberg recruited a proportionally stratified random sample of patients with cancer and survivors of cancer based on a full census. RESULTS: In total, 838 participants completed the survey. Approximately all participants (810/838, 96.7%) showed general willingness to make clinical data available for biomedical research purposes; however, they expected certain requirements to be met, such as comparable data protection standards for data use abroad and the possibility to renew consent at regular time intervals. Most participants (620/838, 73.9%) supported data use also by researchers in commercial companies. More than half of the participants (503/838, 60%) were willing to give up control over clinical data in favor of research benefits. Most participants expressed acceptance of the broad consent model (494/838, 58.9%), followed by data use by default (with the option to opt out at any time; 419/838, 50%); specific consent for every study showed the lowest acceptance rate (327/838, 39%). Patients expected physicians to share their data (763/838, 91.1%) and their fellow patients to support secondary use with their clinical data (679/838, 81%). CONCLUSIONS: Although patients' general willingness to make their clinical data available for biomedical research purposes is very high, the willingness of a substantial proportion of patients depends on additional requirements. Taking these perspectives into account is essential for designing trustworthy governance of clinical data reuse and sharing. The willingness to accept the loss of control over clinical data to enhance the benefits of research should be given special consideration.

The international data governance landscape.

As the adoption of digital health accelerates health research increasingly relies on large quantities of biomedical data. Research institutions scattered across a large number of jurisdictions collaborate in producing and analyzing biomedical big data. National data protection legislation, for its part, grows increasingly complex and localized. To respond to heterogeneous legal requirements arising in numerous jurisdictions, decentralized health consortia must develop scalable organizational and 6 technological arrangements that enable data flows across jurisdictional boundaries. In this article, proposals are made to enable health sector organisations to align established biomedical ethics process and data analysis practices to shifting data protection norms through both public law co-regulation, private law tools, and design-oriented approaches.

Longitudinal Health Studies: Secondary Uses Serving the Future.

Our research compares the ethical and institutional conditions that govern the sharing and secondary use of longitudinal population health data from multiple cohorts. The data use and data sharing conditions applicable to 27 population health cohorts were assessed. This assessment was performed in consulting the informed consent materials and institutional policies applicable to the use of data. Descriptions drawn from the research ethics consent materials were refined through dialog with the institutional staff responsible for overseeing access to data, where possible. Our results demonstrate that data of longitudinal population health cohorts assessed can generally be shared and used for secondary purposes. However, the purposes of secondary use and the preconditions applicable thereto are highly variable. Heterogeneous use conditions can also impede the storage of legacy research data and the pooling thereof for the purpose of common reuse.

Consent recommendations for research and international data sharing involving persons with dementia.

Consent is generally required for research and sharing rich individual-level data but presents additional ethical and legal challenges where participants have diminished decision-making capacity. We formed a multi-disciplinary team to develop best practices for consent in data-intensive dementia research. We recommend that consent processes for research and data sharing support decision-making by persons with dementia, protect them from exploitation, and promote the common good. Broad consent designed to endure beyond a loss of capacity and combined with ongoing oversight can best achieve these goals. Persons with dementia should be supported to make decisions and enabled to express their will and preferences about participation in advance of a loss of capacity. Regulatory frameworks should clarify who can act as a representative for research decisions. By promoting harmonization of consent practices across institutions, sectors, and countries, we hope to facilitate data sharing to accelerate progress in dementia research, care, and prevention.

SpainUDP: The Spanish Undiagnosed Rare Diseases Program.

One of the IRDiRC goals for 2017⁻2027 is to achieve definitive diagnosis for rare undiagnosed diseases within one year, as delay in diagnosis remains one of the pending issues in the rare diseases field. The Spanish Undiagnosed Rare Diseases Program (SpainUDP) was created in response to this challenging scenario to cover patients' needs and after seeing the success of the Undiagnosed Diseases Program (UDP) in the USA. SpainUDP offers a multidisciplinary approach to those patients who have long sought a diagnosis without any success. During the first phase of the protocol, undiagnosed cases are sent to SpainUDP by individual patients or families, patient organizations or hospitals. After careful analysis of phenotype, data from sequencing experiments (WES) is processed with a standard pipeline and detailed standardized phenotypic information (mapped to the Human Phenotype Ontology, HPO) is connected to genetic data. In addition, the participation of SpainUDP in international initiatives such as the European projects RD-Connect and Solve RD, the Undiagnosed Diseases Network International (UDNI), and the MatchMaker Exchange (MME) platform, allows the establishment of a global data sharing strategy across multiple projects submitting data to these international initiatives. From the official beginning of the program (at the end of 2015) until early 2018, 147 cases were accepted in SpainUDP. During this time, 37 cases (25%) dropped out the program due to several reasons. The remaining 110 cases are distributed as follows: phenotypic and genotypic (WES) characterization was finished in 30 cases, of which 20 (67%) were diagnosed; 21 cases are pending on variants' validation by Sanger sequencing; in 25 cases, WES is ongoing and 34 cases are being studied for deep phenotypic characterization. In conclusion, SpainUDP aims to achieve a diagnosis following two recommendations of the IRDiRC: the patients' diagnosis in as short a time as possible and the promotion of data sharing (especially genomic) at the international level.