Co-designing genomics research with a large group of donor-conceived siblings

Background Human genomics research is growing rapidly. More effective methods are required for co-design and involving people, especially those sub-populations which are inherently high interest to medical research and thus at greater risk of being exploited. This case study documents how we worked with a large group of donor-conceived siblings who share the same sperm donor father, to explore how they might want to engage with and influence any future genomic research. Method A participatory action research process was used to explore the views of a group of 18 people who knew they are donor-conceived siblings. They are part of a larger group of up to 1000 people who share the same sperm donor father but the only ones in contact with each other; it is likely that many of the uncontacted siblings are unaware of their biological father, have been unable to trace others or have died. The discussion explored views about how the group would like to be involved in future research. Five members participated in co-design; 12 completed a pre-discussion online survey; and six participated in an online discussion forum and evaluation survey. The online discussion was led by one facilitator, supported by the study team. Results Of the 18 siblings approached in 2018, 14 participated in the co-design stages or the surveys and online discussion. Co-design informed the research process. Participants reported enjoying the overall experience of the surveys and discussion forum, which were perceived as inclusive and flexible. Most participants’ views regarding the value of involvement in research changed during the process, and ‘widened’ about who should be involved. Participants were supportive of future research being done with the siblings group. All who completed the final survey requested to remain part of the co-design process. Other themes in the online discussion included concerns about conflicting interests and a desire for research participation to improve the situation for people affected by assisted conception. The process informed later discussions in the sibling group about participating in a self-managed biobank and informed decision making about participating in genomics research. Conclusion Findings from this study help inform ways in which people from certain sub-populations can be involved in planning and defining their participation in genomic research, particularly those that are inherently high interest to medical research and thus at greater risk of exploitation. This process provides a replicable method of involving potential participants in co-designing genomics research using online discussions, with positive outcomes. Reporting this study using ‘Standardised data on initiatives (STARDIT)’ to report the process allows comparison with other studies. Supplementary Information The online version contains supplementary material available at 10.1186/s40900-021-00325-7.

GRIPP2 report for 'Involving people affected by a rare condition in shaping future genomic research' This report has been completed using the 'GRIPP2 reporting checklists: tools to improve reporting of patient and public involvement in research' available at https://doi.org/10.1136/bmj.j3453.

1: Aim Report the aim of PPI in the study
Participatory action research to involve members of a sibling group in online discussions about how they would like to be involved in future research.

2: Methods
Provide a clear description of the methods used for PPI in the study The research process was co-designed using a participatory action research method to involve people from the sibling group in the co-design of online discussions to explore future genomic research with members of the group. Participants were also involved in checking the final version of the paper.

3: Study results
Outcomes-Report the results of PPI in the study, including both positive and negative outcomes Improved participant information resources, improved wording that was culturally appropriate (using terminology preferred by the sibling group to describe biological relations), improved online discussion, improved learning resources for participants, improved co-design process.
Enablers of involvement: Four participants reported specific things about the way this study was conducted that enabled their involvement. One participant said the entire process was 'assiduous' and that the 'intent of this project' was 'obviously thoughtful and interesting'[P9]. One participant said the 'system seemed to work well' [P7]. Another added that being used to online platforms like Loomio, or having previous experience of similar platforms and 'used to' that way of communicating might facilitate involvement using that communication mode. One participant

Section and topic Category description Data
suggested an alternative discussion format where the participants discussed a thread for 2 days and then had a 3 day break before coming to another thread [P7]. The Facilitator (MC) stated that they felt more time was required in the co-design process.

4: Discussion and conclusions
Outcomes-Comment on the extent to which PPI influenced the study overall. Describe positive and negative effects Involvement improved participant information resources, improved wording that was culturally appropriate (using terminology preferred by the group to describe themselves), improved online discussion, improved learning resources for participants, improved co-design process.

Section and topic Category description Data
Involving potential participants in co-defining language used to describe the group of people affected helped ensure that language was acceptable and appropriate.
Involving participants in co-designing the research process resulted in a number of changes to the study design, including improving language used in recruitment and learning resources.
The process of involving people can be viewed as a learning experience for both the participants involved and study team members. The process changed participants' views about who should be involved, which can be viewed as an impact of 'transformative learning'.

5:
Reflections/critical perspective Comment critically on the study, reflecting on the things that went well and those that did not, so others can learn from this experience The co-design process took longer than expected owing to ethical 'grey areas' with no clear instruction on whether ethics approval was required to involve people in co-design. As a result an ethics application was made and subsequent feedback from the co-design process was integrated using modifications to the ethics application.
Involving potential participants in co-defining language used to describe the group of people affected helped ensure that language was acceptable and appropriate.
Involving participants in co-designing the research process resulted in a number of changes to the study design, including improving language used in recruitment and learning resources.
The process of involving people can be viewed as a learning experience for both the participants involved and study team members. The process changed participants' views about who should be involved, which can be viewed as an impact of 'transformative learning'.