Managing misaligned paternity findings in research including sickle cell disease screening in Kenya: ‘Consulting communities’ to inform policy

Abstract

The management of misaligned paternity findings raises important controversy worldwide. It has mainly, however, been discussed in the context of high-income countries. Genetic and genomics research, with the potential to show misaligned paternity, are becoming increasingly common in Africa. During a genomics study in Kenya, a dilemma arose over testing and sharing information on paternal sickle cell disease status. This dilemma may be paradigmatic of challenges in sharing misaligned paternity findings in many research and health care settings. Using a deliberative approach to community consultation to inform research practice, we explored residents' views on paternal testing and sharing misaligned paternity information. Between December 2009 and November 2010, 63 residents in Kilifi County were engaged in informed deliberative small group discussions, structured to support normative reflection within the groups, with purposive selection to explore diversity. Analysis was based on a modified framework analysis approach, drawing on relevant social science and bioethics literature.The methods generated in-depth individual and group reflection on morally important issues and uncovered wide diversity in views and values. Fundamental and conflicting values emerged around the importance of family interests and openness, underpinned by disagreement on the moral implications of marital infidelity and withholding truth. Wider consideration of ethical issues emerging in these debates supports locally-held reasoning that paternal sickle cell testing should not be undertaken in this context, in contrast to views that testing should be done with or without the disclosure of misaligned paternity information. The findings highlight the importance of facilitating wider testing of family members of affected children, contingent on the development and implementation of national policies for the management of this inherited disorder. Their richness also illustrates the potential for the approach adopted in this study to strengthen community consultation.

Community engagement strategies for genomic studies in Africa: a review of the literature

“…Tedrow et al used a qualitative multi-case design [ 39 ] while Tindana et al used focus group discussions and in-depth interviews with specific groups in the community to consider how CE enhanced research understanding and the informed consent process [ 10 ]. Marsh et al used action research methods (analysis of documentation and observations) including qualitative and quantitative content analysis and thematic analysis to develop greater understanding about the strengths and challenges of community engagement in supporting ethical research practice [ 46 ]. Morin et al used a rapid assessment model of data collection to examine the process of CAB development and the interaction between CAB members, research team members and research participants [ 22 ].…”

“…In the context of genomic research, the literature suggests that community engagement can assist in identifying and clarifying community members’ misunderstanding about the aims and intentions of the research project, as well as concerns about the procedures used to obtain data and samples [ 11 ]. It can also provide an opportunity for the research team to take into account the opinions of both the staff and community relating to issues pertinent to the study, allowing for adapting and modifying of information, messages and research methods [ 18 , 19 , 46 ].…”

Do solidarity and reciprocity obligations compel African researchers to feedback individual genetic results in genomics research?

“…Holding the dFGDs in two separate stages was intended to: (1) create enough time for engagement with participants and support information sharing especially because we were investigating a topic with which participants were unfamiliar; (2) provide participants with an opportunity to reflect on the issue being studied in-between the discussions; and (3) allow revisiting of views over time [ 36 , 37 ]. Whilst we interrogated a broader range of issues relating to the return of individual genetic research results, we also specifically probed solidarity and reciprocity, for instance in relation to questions about why participants thought that individual genetic results should be returned, whether, how and why participants should be appreciated for their participation and who they thought was responsible for covering costs of follow-up care and why.…”

Deliberation to Promote Shared Sovereignty in Health Research: Four Questions to Clarify Goals, Methods, and Scope

“…There are several research groups that have sought to conduct deliberation of the standard Pratt and Hyder seek to achieve (Burgess, 2014; Longstaff & Burgess, 2010; V. Marsh et al, 2013).…”

“…But such deliberation is resource and time intensive and limited in scope. For instance, in the examples we know of, deliberation was used to inform the development of a governance framework for biobanks (O’Doherty et al, 2011) or to seek community feedback on the return of individual genetic research results (Marsh, Kombe, Fitzpatrick, Molyneux, & Parker, 2013), but it was not used to also inform on priority setting, resource allocation decisions, and the other aspects of research collaboration identified by Pratt and Hyder. There is no reason, of course, that the deliberative panels forwarded by Burgess et al could not also be convened to explore those questions – but this may have implications for panel composition for instance.…”