Original article
Informed consent for enrolling minors in genetic susceptibility research: a qualitative study of at-risk children’s and parents’ views about children’s role in decision-making

https://doi.org/10.1016/S1054-139X(02)00459-7Get rights and content

Abstract

Purpose

To better understand the process by which families at increased risk of disease would decide in order to enroll their children in genetic susceptibility research to develop recommendations regarding the informed consent process by which at-risk children are enrolled in such research in the future.

Methods

Parents and children (ages 10–17 years) from families at increased risk for heart disease (n = 21 dyads) or breast cancer (n = 16 dyads) participated in two face-to-face, audio-taped, semi-structured interviews: Initial interviews were conducted with parents and children separately, and follow-up family interviews were conducted 1 year later. Interview transcripts were coded based on common themes.

Results

Families vary in the stage at which, and degree to which, children would be involved in decision-making about research participation. In general, the older/more mature the child, the less risky the research and the more open the communication style, the greater the likelihood that decisions would be made jointly. Most children wanted some parental input, but still thought the final decision should be theirs. Most parents would want to make the initial decision about whether it would be reasonable to consider enrolling their child in the research being proposed, but none opposed the child having some time alone with the researcher. All parents and children in our study placed extreme importance on not forcing children to participate in nontherapeutic research if they do not want to.

Conclusions

Decision-making about enrolling children in genetic susceptibility research should be based on an informed consent process that (a) gives parents and children sufficient opportunity to ask questions of the researcher(s) and to communicate with one another, and (b) gives children the opportunity to exercise their right to refuse participation without parental influence. This process should be tailored to the child’s maturity level and style of communication in the family.

Section snippets

Methods

We chose a qualitative approach for this study for several reasons. An open-ended format allowed us to: (a) explore the full range of perceptions and beliefs regarding the involvement of minors in genetic susceptibility research, (b) observe the evolution of individuals’ perceptions in the context of an interactive exchange, and (c) elicit detailed descriptions of family dynamics and decision-making patterns. In developing a methodology that best addressed our specific set of research

Results

Thirty-seven dyads of parents and children participated in the initial interviews. For the follow-up interviews, 20 families participated. Two families declined participation in the follow-up interview and 15 families did not respond or were lost to follow-up. Table 2 provides the distribution of our sample by type of disease, age of child, and race. As shown, we were unable to recruit a sufficient number of African-American families to determine whether there were any racial differences in

Discussion

The families that participated in our study were highly variable in the degree to which children would be involved in decision-making about research participation. Most families used criteria such as developmental stage of the child and degree of risk of the research to determine the respective roles that each would play in decision-making. In general, the older/more mature the child, and the less risky the research, the more likely decisions would be made jointly.

Although we did not involve

Acknowledgements

We are indebted to the parents and children who participated in this study. We are also grateful to our colleagues, at both University of Virginia (e.g., Susan Meisfeldt) and Johns Hopkins (e.g., Lillie Shockney, Roger Blumenthal, M.D.), who assisted us in recruiting these families. We also appreciate the help of our interviewers, Jessica Rispoli and Dana Wollins, and our Research Assistant, Jennifer Langer. Neil A. Holtzman, M.D., M.P.H. and Peter O. Kwiterovich, M.D. were co-investigators on

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    This work was supported by a grant (R01 HD36189-01) from the National Institute of Child Health and Development (NICHD), and an Administrative Supplement from the National Human Genome Research Institute (NHGRI).

    The full text of this article is available via JAH online at http://www.elsevier.com/locate/jahonline.

    View full text