Handling ethical, legal and social issues in birth cohort studies involving genetic research: responses from studies in six countries

Abstract

BackgroundResearch involving minors has been the subject of much ethical debate. The growing number of longitudinal, pediatric studies that involve genetic research present even more complex challenges to ensure appropriate protection of children and families as research participants. Long-term studies with a genetic component involve collection, retention and use of biological samples and personal information over many years. Cohort studies may be established to study specific conditions (e.g. autism, asthma) or may have a broad aim to research a range of factors that influence the health and development of children. Studies are increasingly intended to serve as research platforms by providing access to data and biological samples to researchers over many years.This study examines how six birth cohort studies in North America and Europe that involve genetic research handle key ethical, legal and social (ELS) issues: recruitment, especially parental authority to include a child in research; initial parental consent and subsequent assent and/or consent from the maturing child; withdrawal; confidentiality and sample/data protection; handling sensitive information; and disclosure of results.MethodsSemi-structured telephone interviews were carried out in 2008/09 with investigators involved in six birth cohort studies in Canada, Denmark, England, France, the Netherlands and the United States. Interviewees self-identified as being knowledgeable about ELS aspects of the study. Interviews were conducted in English.ResultsThe studies vary in breadth of initial consent, but none adopt a blanket consent for future use of samples/data. Ethics review of new studies is a common requirement. Studies that follow children past early childhood recognise a need to seek assent/consent as the child matures. All studies limit access to identifiable data and advise participants of the right to withdraw. The clearest differences among studies concern handling of sensitive information and return of results. In all studies, signs of child abuse require reports to authorities, but this disclosure duty is not always stated in consent materials. Studies vary in whether they will return to participants results of routine tests/measures, but none inform participants about findings with unknown clinical significance.ConclusionsAnalysis of how cohort studies in various jurisdictions handle key ELS issues provides informative data for comparison and contrast. Consideration of these and other examples and further scholarly exploration of ELS issues provides insight on how best to address these aspects in ways that respect the well-being of participants, especially children who become research subjects at the start of their lives.

Children, biobanks and the scope of parental consent

“…One could argue, however, that that this is not really blanket consent, as the specific context of these studies allow for frequent contact between researchers, children and parents, thereby, providing many opportunities to reconsent. 24 An interview-based study with professionals associated with pediatric biobanks has shown that they would prefer 'broad consent' , but also think that if the research takes a too big step away from the original protocols, ethics committees should decide whether re-consent is necessary. 7 These studies suggest a discrepancy between what is formally consented (which is fairly broad and includes any potential future research) and the actual practices researchers feel comfortable with.…”

Clarifying assent in pediatric research

“…[21][22][23][24][25][26][27][28] The importance of conducting ethically sound pediatric biobank research 29 and the rapid developments in this field stress the need to scrutinize the concept of assent in pediatric biobank research. 14,18 Here, we will provide a conceptual analysis of assent, discuss the different underlying ethical principles and its, in our opinion, most tenable interpretation in pediatric research. In addition, we will consider how it should be implemented in biomedical research.…”

“…12 With regard to the different types of pediatric biobanks, the prevailing view is that assent should be obtained when possible. 3,[13][14][15][16][17][18][19] However, though many emphasize the importance of assent, few explain how they understand the concept and few have elaborated on the underlying grounds of assent and its role in pediatric biobanks. 20 This is not surprising as no consensus exists about how assent should be interpreted and implemented in biomedical research.…”

Ethics of children’s participation in a Saudi biobank: an exploratory survey

“…2,3 One distinct challenge is related to a child's ability to make a decision with regard to participating in medical research and sharing the information. There has been much discussion about the validity of parental consent and/or child assent, 4-6 the most suitable age for accepting child consent, 7 child withdrawal, 2,6,8,9 and reconsent when a child reaches adulthood. 2 Questions also have been asked about the right to share a child's information and whether there is any difference between medical and genetic information in relation to confidentiality.…”