Ethical, Legal, and Social Issues (ELSI) of Responsible Data Sharing Involving Children in Genomics: A Systematic Literature Review of Reasons

Abstract

Background: Progress in precision medicine relies on the access to, use of, and exchange of genomic and associated clinical data, including from children. The ethical, legal, and social issues (ELSI) of such data access, use, and exchange may be accentuated in the pediatric context due in part to the highly sensitive nature of genomic data, children's consentrelated vulnerabilities, and uncertain risks of reidentification. Systematic analyses of the ELSI and scientific reasons for why and how genomic data may be shared responsibly are, however, limited. Methods: We conducted a modified systematic review of reasons according to Sofaer and Strech to examine the ELSI and scientific reasons for "responsible" sharing of children's genomic and associated clinical data. Empirical articles, commentaries, and datasharing policies indexed in Medline, Scopus, Web of Science, and BIOSIS were included in the analysis if they discussed ELSI and were published between 2003 and 2017 in English. Results: One hundred and fifty-one records met our inclusion criteria. We identified 11 unique reasons and 8 subreasons for why children's genomic data should or should not be shared. Enhancing the prospect of direct and indirect benefits and maximizing the utility of children's data were top reasons why data should be shared. Inadequate data privacy protection was the leading reason why it should not. We furthermore identified 8 reasons and 30 subreasons that support conditional data sharing, in which recontact for the continued use of children's data once they reach the age of majority was the most frequently endorsed condition. Conclusions: The complete list of ELSI reasons and responsible conditions provides an evidentiary basis upon which institutions can develop data-sharing policies. Institutions should encourage the sharing of children's data to advance genomic research, while heeding special reconsent and data protection mechanisms that may help mitigate uncertain longitudinal risks for children and families.

Genomic answers for children: Dynamic analyses of >1000 pediatric rare disease genomes

“…Greater data sharing is paramount for enhancing benefits to participants and advancing scientific progress, along with maximizing the utility of genomic data. 37 Unfortunately, hesitancy toward extensive data sharing persists among investigators because of reasons that include the arduous processes required for data sharing, concerns about participant privacy, and fear of loss of priority in data publication. 37,38 Our study follows regulations and considers recommendations for responsible sharing of pediatric genomic data to support the benefits of data sharing to research participants and patients while protecting privacy.…”

“…37,38 Our study follows regulations and considers recommendations for responsible sharing of pediatric genomic data to support the benefits of data sharing to research participants and patients while protecting privacy. 37…”

IGenomic answers for children: Dynamic analyses of >1000 pediatric rare disease genomes

“…Greater data sharing is paramount for enhancing benefits to participants and advancing scientific progress, along with maximizing the utility of genomic data. 40 Unfortunately, hesitancy towards extensive data sharing persists among investigators due to reasons that include the arduous processes required for data sharing, concerns about participant privacy, and fear for loss of priority in data publication. 40,41 Our study follows regulations and considers recommendations for responsible sharing of pediatric genomic data to support the benefits of data sharing to research participants and patients while protecting privacy.…”

“…40,41 Our study follows regulations and considers recommendations for responsible sharing of pediatric genomic data to support the benefits of data sharing to research participants and patients while protecting privacy. 40…”

Sharing and Safeguarding Pediatric Data

“…However, minors are very often excluded from data sharing initiatives, especially where genomic data is involved ( Rahimzadeh et al, 2018 ). The sensitivity of genomic data, consent-related issues, and the uncertain risk of re-identification have all been raised as reasons for excluding minors from data sharing ( Rahimzadeh et al, 2020 ). International research ethics guidelines recognize the unique vulnerability of minors as research participants and the requirement for the implementation of special protections for minors (e.g.…”