Attitudes of stakeholders in psychiatry towards the inclusion of children in genomic research

Abstract

BackgroundGenomic sequencing of children in research raises complex ethical issues. This study aims to gain more knowledge on the attitudes towards the inclusion of children as research subjects in genomic research and towards the disclosure of pertinent and incidental findings to the parents and the child.MethodsQualitative data were collected from interviews with a wide range of informants: experts engaged in genomic research, clinical geneticists, persons with mental disorders, relatives, and blood donors. Quantitative data were collected from a cross-sectional web-based survey among 1227 parents and 1406 non-parents who were potential stakeholders in psychiatric genomic research.ResultsParticipants generally expressed positive views on children’s participation in genomic research. The informants in the qualitative interviews highlighted the age of the child as a critical aspect when disclosing genetic information. Other important aspects were the child’s right to an autonomous choice, the emotional burden of knowing imposed on both the child and the parents, and the possibility of receiving beneficial clinical information regarding the future health of the child. Nevertheless, there was no consensus whether the parent or the child should receive the findings. A majority of survey stakeholders agreed that children should be able to participate in genomic research. The majority agreed that both pertinent and incidental findings should be returned to the parents and to the child when of legal age. Having children does not affect the stakeholder’s attitudes towards the inclusion of children as research subjects in genomic research.ConclusionOur findings illustrate that both the child’s right to autonomy and the parents’ interest to be informed are important factors that are found valuable by the participants. In future guidelines governing children as subjects in genomic research, it would thus be essential to incorporate the child’s right to an open future, including the right to receive information on adult-onset genetic disorders.

Psychiatric genomics researchers’ perspectives on best practices for returning results to individual participants

“…Despite increasing consensus that some clinically relevant findings should be offered to individual participants in genomics research, 6,8,[16][17][18] including psychiatric genetics, 10,[19][20][21] there is little empirical data about how to ideally manage return of results (RoR) in the psychiatric genetics research context. 22,23 Most research on RoR in genetics research has focused on whether results should be offered, and the perceived risks and benefits of returning results, [7][8][9]18,24,25 along with which types of results (e.g., medically actionable, clinically valid but not actionable) are justifiable and recommended to return.…”

“…7,10 While these studies have opened a broader dialogue about whether results should be offered and why, only a few empirical studies to date have specifically addressed which results should be offered and how results should be returned in psychiatric genetics research. [19][20][21]27 No empirical study has examined these issue from the perspective of psychiatric genomics researchers who are key stakeholders in implementing plans for returning results.…”

Psychiatric genetics researchers' views on offering return of results to individual participants

“…Likewise, Sundby et al () found that psychiatric genetics stakeholders (i.e., patients with mental health disorders, their relatives, and clinicians) favor the return of both pertinent (95%) and incidental (91%) findings to research participants (Sundby et al, ), including to parents of child participants and to the children themselves when of legal age (Sundby et al, ). In another study, 97% of patients with a psychiatric disorder and unaffected relatives reported that they would like to be informed of any clinically relevant genomic findings generated in a study (Bui, Anderson, Kassem, & McMahon, ).…”

Attitudes toward the right to autonomous decision‐making in psychiatric genetic testing: Controversial and context‐dependent

“…Available guidelines recommend that predictive genetic testing should only be performed in minors if there is a potential direct medical benefit, or if preventive measures initiated in childhood will decrease morbidity and mortality (Committee on Bioethics, 2013; EURAT, ; European Society of Human Genetics, ). However, some experts question this approach, and suggest that it may be too narrow (Hardart & Chung, ; Sundby et al, ). The findings of some studies suggest that parents may be more reluctant to consent to genetic testing in their children following in‐depth discussion of the potential consequences (Bernhardt, Tambor, Fraser, Wissow, & Geller, ; Geller, Tambor, Bernhardt, Wissow, & Fraser, ).…”