An Observational Study of Children’s Involvement in Informed Consent for Exome Sequencing Research

Abstract

The goal of this study was to examine children’s involvement in consent sessions for exome sequencing research and associations of involvement with provider and parent communication. Participants included 44 children (8–17 years) from five cohorts who were offered participation in an exome sequencing study. The consent sessions were audiotaped, transcribed, and coded. Providers attempted to facilitate the child’s involvement in the majority (73%) of sessions, and most (75%) children also verbally participated. Provider facilitation was strongly associated with likelihood of child participation. These findings underscore that strategies such as asking for children’s opinions and soliciting their questions show respect for children and may increase the likelihood that they are engaged and involved in decisions about research participation.

How Do Providers Discuss the Results of Pediatric Exome Sequencing with Families?

“…Pediatric participants, who were present in about half of the sessions analyzed, may be especially confused when results are returned in such a highly technical manner. Tailored strategies will likely be needed to engage them in these discussions and ensure they have a developmentally appropriate understanding of the findings and implications for their health care [22,23]. …”

“…A detailed description of subject recruitment and informed consent process is published elsewhere [22,23]. Briefly, clinicians from five disease-specific clinics providing care to children from the study cohorts recruited participants during an initial or follow-up clinic visit.…”

Informed Consent in Social Sciences Research: Ethical Challenges

“…It is relevant in the way of proceeding in the respect for the intrinsic and extrinsic values of science, both during the process and in the development and dissemination of the final product(s). Thus, there is no ethically neutral research, and ethics goes through all phases of scientific research, whatever its audience (Gómez, Flores, & Jimé nez, 1999;Tuckman, 2000;Lima, 2006;Graue & Walsh, 2003;Miller, Werner-Lin, Walser, Biswas, & Bernhardt, 2017), without forgetting, naturally, the new ethical challenges posed by the Internet and the digital world (Peixoto, 2017;Saunders, Kitzinger, & Kitzinger, 2015).…”

“…142 and 143), that the following elements are present: identification of the researcher, explanation of the objectives and procedures to be used, information on the probable length of the participation, indication of possible risks for participating, information on whether or not compensation for possible damages is available, description of expected benefits resulting from the research, indication of voluntary participation and of the possibility of the respondent's withdrawal at any time, indication of procedures to preserve confidentiality of information, indication of how data will be stored and who will have access to them, indication of people to contact throughout and at the end of the research in case of need, and indication of the entities that promote the research and its context. The need to obtain this informed consent is even more pressing in situations where participants are minors and/or not fully aware of the implications of their participation, such as children (Miller et al, 2017): It is critical for investigators and individuals conducting informed consent with families to understand that there are multiple strategies to show respect for children and encourage their active involvement in consent discussions. IRBs should consider educational initiatives, such as training sessions and toolkits, to increase awareness and provide specific guidance regarding communication strategies to increase child participation in informed consent sessions in general, and in decision making about exome sequencing research with disclosure of secondary findings in particular (p. 12).…”

Challenges to informed consent for exome sequencing: A best–worst scaling experiment

“…The challenges to consent for exome sequencing are particularly salient in the pediatric setting where children undergoing a diagnostic workup have the potential to learn information about risk for unrelated adult onset conditions that could influence developing self‐identity (Miller, Werner‐Lin, Walser, Biswas, & Bernhardt, ; Werner‐Lin, Tomlinson, Miller, & Bernhardt, ). The decision to opt out of receiving medically actionable secondary findings should be made prior to undergoing testing, and parents who have difficulty grasping the potential consequences of the results may report lower confidence in their ability to make an informed choice (O’Daniel et al, ; Turbitt et al, ).…”