International Data Sharing and Rare Disease: The Importance of Ethics and Patient Involvement

Thorogood, Adrian

doi:10.5772/intechopen.91237

Cited by 10 publications

(10 citation statements)

References 28 publications

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“…Over the past years, NASA's Life Sciences Data Archive (LSDA), PubSpace, NASA NTRS as well as GeneLab initiatives, have sought to improve data availability and thus can be regarded as a great leap forward in working with sensitive data 74,75 Primarily given the wide array of rare diseases and low incidence rate, it is highly unlikely for a single group to advance research alone 77 . Data sharing may be a valuable resource in understanding natural history, disease progression, and providing an adequate sample size to work with.…”

Section: Tailored Approachesmentioning

confidence: 99%

“…Data privacy laws are increasing in sophistication globally to allow for a transition into the big data era of mass data sharing, often resulting in tightened laws with higher consent standards and safeguards in place, allowing for less flexibility to share samples and data internationally 77 . In order to achieve a balance between too lenient and too rigid privacy laws, increased patient involvement in research design, security protections and transparency about how data sharing occurs have been recommended 48,77 . The extent of the experimental information collected and the degree of privacy that's regulated will remain a topic that requires further discussion among stakeholders in both communities.…”

Section: Tailored Approachesmentioning

confidence: 99%

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Rare diseases and space health: optimizing synergies from scientific questions to care

et al. 2022

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Knowledge transfer among research disciplines can lead to substantial research progress. At first glance, astronaut health and rare diseases may be seen as having little common ground for such an exchange. However, deleterious health conditions linked to human space exploration may well be considered as a narrow sub-category of rare diseases. Here, we compare and contrast research and healthcare in the contexts of rare diseases and space health and identify common barriers and avenues of improvement. The prevalent genetic basis of most rare disorders contrasts sharply with the occupational considerations required to sustain human health in space. Nevertheless small sample sizes and large knowledge gaps in natural history are examples of the parallel challenges for research and clinical care in the context of both rare diseases and space health. The two areas also face the simultaneous challenges of evidence scarcity and the pressure to deliver therapeutic solutions, mandating expeditious translation of research knowledge into clinical care. Sharing best practices between these fields, including increasing participant involvement in all stages of research and ethical sharing of standardized data, has the potential to contribute to humankind’s efforts to explore ever further into space while caring for people on Earth in a more inclusive fashion.

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Section: Tailored Approachesmentioning

confidence: 99%

Section: Tailored Approachesmentioning

confidence: 99%

Rare diseases and space health: optimizing synergies from scientific questions to care

et al. 2022

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“…Intellectual property rights and considerations surrounding human data disclosure pose consistent challenges (7,8). Though data anonymization has been advanced as a solution, risks of re-identification remain, especially among vulnerable populations (9,10). Additionally, the sharing of anonymized data may not always be covered by the original consent agreements, adding a layer of complexity to privacy protection.…”

Section: Introductionmentioning

confidence: 99%

A Decade of Progress: Insights of Open Data Practices in Biosciences at the University of Edinburgh

Deeb,

Wong,

Usman

et al. 2024

Preprint

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The evolution of research practices emphasizes the role of Open Data in fostering transparency and collaboration. This study evaluates the adoption of Open Data practices in the School of Biological Sciences at a research-intensive university in the United Kingdom. Our analysis of research data sharing from 2014 to 2022 manually reviewed 193 journal articles against criteria for Openness and FAIRness, including the Completeness of data shared relative to data generated. The findings reveal an uptick in data completeness and reusability, with a clear influence of data type, and genomic data being shared more frequently than image data. Data availability statements (DAS) and preprint sharing show a strong correlation with higher Openness and FAIRness scores. Since 2016, when the FAIR guidelines were published, data Reusability increased along with the inclusion of Data Availability Statements. On the other hand, since the COVID-19 pandemic, we have found a substantial increase in preprint sharing and significant improvements in Completeness, Reusability, and Accessibility scores. This paper documents a local research institute's journey towards Open Data, addressing the changes and advocating for best practices to nurture this progression.

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“…However, this type of research requires a combination of genetic and phenotypic information which presents a high privacy risk for these patients and their relatives. Assessing rare disease patients and carers' views about involvement in decision-making concerned with their data can help in designing a data governance structure suitable to meet their needs and expectations from biomedical and health care research and to enhance the trustworthiness of institutions involved in research ( 16 , 43 ).…”

Section: Introductionmentioning

confidence: 99%

Public Views About Involvement in Decision-Making on Health Data Sharing, Access, Use and Reuse: The Importance of Trust in Science and Other Institutions

Nwebonyi

Silva

Freitas

2022

Front. Public Health

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BackgroundData-intensive and needs-driven research can deliver substantial health benefits. However, concerns with privacy loss, undisclosed surveillance, and discrimination are on the rise due to mounting data breaches. This can undermine the trustworthiness of data processing institutions and reduce people's willingness to share their data. Involving the public in health data governance can help to address this problem by imbuing data processing frameworks with societal values. This study assesses public views about involvement in individual-level decisions concerned with health data and their association with trust in science and other institutions.MethodsCross-sectional study with 162 patients and 489 informal carers followed at two reference centers for rare diseases in an academic hospital in Portugal (June 2019–March 2020). Participants rated the importance of involvement in decision-making concerning health data sharing, access, use, and reuse from “not important” to “very important”. Its association with sociodemographic characteristics, interpersonal trust, trust in national and international institutions, and the importance of trust in research teams and host institutions was tested.ResultsMost participants perceived involvement in decision-making about data sharing (85.1%), access (87.1%), use (85%) and reuse (79.9%) to be important or very important. Participants who ascribed a high degree of importance to trust in research host institutions were significantly more likely to value involvement in such decisions. A similar position was expressed by participants who valued trust in research teams for data sharing, access, and use. Participants with low levels of trust in national and international institutions and with lower levels of education attributed less importance to being involved in decisions about data use.ConclusionThe high value attributed by participants to involvement in individual-level data governance stresses the need to broaden opportunities for public participation in health data decision-making, namely by introducing a meta consent approach. The important role played by trust in science and in other institutions in shaping participants' views about involvement highlights the relevance of pairing such a meta consent approach with the provision of transparent information about the implications of data sharing, the resources needed to make informed choices and the development of harm mitigation tools and redress.

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International Data Sharing and Rare Disease: The Importance of Ethics and Patient Involvement

Cited by 10 publications

References 28 publications

Rare diseases and space health: optimizing synergies from scientific questions to care

Rare diseases and space health: optimizing synergies from scientific questions to care

A Decade of Progress: Insights of Open Data Practices in Biosciences at the University of Edinburgh

Public Views About Involvement in Decision-Making on Health Data Sharing, Access, Use and Reuse: The Importance of Trust in Science and Other Institutions

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