Growing up as a research subject: ethical and legal issues in birth cohort studies involving genetic research.

Introduction Many population genetic research projects seek to recruit women during pregnancy or infants at birth for longitudinal birth cohort studies that investigate how early environmental conditions interact with a child's genome to increase disease susceptibility or confer protective effects. (1) Researchers worldwide are sleuthing for genetic factors related to major child health problems such as obesity, diabetes, behavioural disorders, allergies and asthma. These research initiatives depend on the availability of biological samples collected periodically from children, their mothers and sometimes other family members--often including samples of blood, placenta, hair, nails and urine--and collection of information about lifestyle, environmental exposures and health. One commentator observes: Increasingly, research involving children uses longitudinal designs to identify the developmental trajectories of environmental health problems and the single or joint health effects of heredity and prenatal and postnatal exposure to environmental agents. By its nature, such research often involves asymptomatic children who will or will not be affected by environmental agents under investigation or who will develop a disorder previously not known to be associated with an environmental agent. (2) Birth cohort studies are notoriously challenging and expensive to design and implement. Indeed, "such studies involve a long-term contact with the participating families where the children, in some cases, will be born and grow up during the project. " (3) This feature contributes to the immense scientific value of cohort studies, but also is the source of many legal and ethical concerns. Infants and young children are incapable of giving consent to participate in health research, so parents or guardians must give their permission for a child to take part. But legal and ethical rules limit parental rights to permit a child to participate in research, particularly research that is of no direct benefit to the child. How do these limits apply to parental permission for an infant to become part of a birth cohort that will follow the child's health and development over many years? As children mature, they develop capacity to make their own choices about whether they wish to continue participating in research. Empirical studies of minors' capacity to consent suggest that psychosocial maturity to understand the nature of research activities and the rights of research subjects develops by age seven to 10, and by age 14 to 16 an adolescent's capacity is generally the same as an adult's. (4) In a cohort study, where a child grows up as a research subject, when--if at all--does a child have a right to express his or her own, independent choice about continuing to participate? Long-term studies involving genetic research also raise special informational privacy risks. Vast amounts of personal information are collected and analysed over the course of a cohort study and genetic research may reveal previously unknown--and possibly unwelcome--information about participants and their biological relatives. Researchers must implement robust confidentiality and security measures to safeguard participants' information and must also develop policies about returning research results to participants. Finally, researchers must be aware of potential obligations to report information acquired in the course of research to third parties, such as concern that a child is at risk of abuse or neglect. This article begins with an overview of the general rights of research participants and the special context of birth cohort studies involving genetic research, then analyses in detail legal and ethical issues related to consent for children to participate in research, risks and perceptions of risk, factors that motivate research participation, conflicts between children and parents over participation in research, rights to withdraw from research and duties to report information acquired during the course of the research, including results of research tests. …