Dynamic consent (DC) is an approach to consent that enables people, through an interactive digital interface, to make granular decisions about their ongoing participation. This approach has been explored within biomedical research, in fields such as biobanking and genomics, where ongoing contact is required with participants. It is posited that DC can enhance decisional autonomy and improve researcher–participant communication. Currently, there is a lack of evidence about the measurable effects of DC-based tools. This article outlines a framework for DC evaluation and reporting. The article draws upon the evidence for enhanced modes of informed consent for research as the basis for a logic model. It outlines how future evaluations of DC should be designed to maximize their quality, replicability, and relevance based on this framework. Finally, the article considers best-practice for reporting studies that assess DC, to enable future research and implementation to build upon the emerging evidence base.

Background: There are increasing calls for patient involvement in sharing health research results, but no evidencebased recommendations to guide such involvement. Our objectives were to: (1) conduct a systematic review of the evidence on patient involvement in results sharing, (2) propose evidence-based recommendations to help maximize benefits and minimize risks of such involvement and (3) conduct this project with patient authors. Methods: To avoid research waste, we verified that no systematic reviews were registered or published on this topic. We co-created, with patients, a PRISMA-P-compliant protocol. We included peer-reviewed publications reporting the effects of patient involvement in preparing peer-reviewed publications or results summaries from health research studies. We searched (9/10/2017) MEDLINE, EMBASE and the Cochrane Database of Systematic Reviews, and secondary information sources (until 11/06/2018). We assessed the risk of bias in eligible publications and extracted data using standardized processes. To evaluate patient involvement in this project, we co-created a Patient Authorship Experience Tool.Results: All nine eligible publications reported on patient involvement in preparing publications; none on preparing results summaries. Evidence quality was moderate. A qualitative synthesis of evidence indicated the benefits of patient involvement may outweigh the risks. We have proposed 21 evidence-based recommendations to help maximize the benefits and minimize the risks when involving patients as authors of peer-reviewed publications. The recommendations focus on practical actions patient and non-patient authors can take before (10 recommendations), during (7 recommendations) and after (4 recommendations) manuscript development. Using the Patient Authorship Experience Tool, both patient and non-patient authors rated their experience highly.Conclusions: Based on a systematic review, we have proposed 21 evidence-based recommendations to help maximize the benefits and minimize the risks of involving patients as authors of peer-reviewed publications.

Background The role of patients in medical research is changing, as more emphasis is being placed on patient involvement, and patient reported outcomes are increasingly contributing to clinical decision-making. Information and communication technology provides new opportunities for patients to actively become involved in research. These trends are particularly noticeable in Europe and the US, but less obvious in Japan. The aim of this study was to investigate the practice of active involvement of patients in medical research in Japan by utilizing a digital platform, and to analyze the outcomes to clarify what specific approaches could be put into practice. Methods We developed the RUDY JAPAN system, an ongoing rare disease medical research platform, in collaboration with the Rare and Undiagnosed Diseases Study (RUDY) project in the UK. After 2 years of preparation, RUDY JAPAN was launched in December 2017. Skeletal muscle channelopathies were initially selected as target diseases, and hereditary angioedema was subsequently added. Several approaches for active patient involvement were designed through patient-researcher collaboration, namely the Steering Committee, questionnaire development, dynamic consent, and other communication strategies. We analyzed our practices and experiences focusing on how each approach affected and contributed to the research project. Results RUDY JAPAN has successfully involved patients in this research project in various ways. While not a part of the initial decision-making phase to launch the project, patients have increasingly been involved since then. A high level of patient involvement was achieved through the Steering Committee, a governance body that has made a major contribution to RUDY JAPAN, and the process of the questionnaire development. The creation of the Patient Network Forum, website and newsletter cultivated dialogue between patients and researchers. The registry itself allowed patient participation through data input and control of data usage through dynamic consent. Conclusions We believe the initial outcomes demonstrate the feasibility and utility of active patient involvement in Japan. The collaboration realized through RUDY JAPAN was enabled by digital technologies. It allowed busy patients and researchers to find the space to meet and work together for the Steering Committee, questionnaire development and various communication activities. While the practice of active patient involvement in Japan is still in its early stages, this research confirms its viability if the right conditions are in place. (331 words).

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Patient involvement (PI) in determining medical research priorities is an important way to ensure that limited research funds are allocated to best serve patients. As a disease area for which research funds are limited, we see a particular utility for PI in priority-setting for medical research on rare diseases. In this review, we argue that PI initiatives are an important form of evidence for policymaking. We conducted a study to identify the extent to which PI initiatives are being conducted in the rare disease field, the features of such initiatives, the trends in the priorities elicited, and the extent to which translation into policy is reported in the academic literature. Here, we report the results of this exploratory review of the English-language literature gathered through online databases and search engines, with the aim of identifying journal articles published prior to December 2020, describing PI initiatives focused on determining priorities for medical research funding in the rare disease field. We identified seven recently-published articles and found that the majority made use of structured methodologies to ensure the robustness of the evidence produced, but found little reported practical implementation or concrete plans for implementation of the results of the initiatives. We conclude that priority-setting initiatives are meaningful mechanisms for involving patients in determining research directions. However, we highlight the importance of translation into policy as a necessary next step to fully utilize the results and move beyond well-intentioned exercises. Finally, we draw attention to the benefits of involving patients throughout this process.