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Genet Med. 2014 Jul;16(7):547-52. doi: 10.1038/gim.2013.190. Epub 2014 Jan 9.

Guidelines for return of research results from pediatric genomic studies: deliberations of the Boston Children's Hospital Gene Partnership Informed Cohort Oversight Board.

Author information

  • 11] Division of Genetics and Genomics, Boston Children's Hospital, Boston, Massachusetts, USA [2] The Manton Center for Orphan Disease Research, Boston Children's Hospital, Boston, Massachusetts, USA [3] Department of Pediatrics, Harvard Medical School, Boston, Massachusetts, USA.
  • 2Division of Genetics and Genomics, Boston Children's Hospital, Boston, Massachusetts, USA.
  • 31] Partners Center for Personalized Genetic Medicine, Boston, Massachusetts, USA [2] Division of Genetics, Department of Medicine, Brigham and Women's Hospital and Harvard Medical School, Boston, Massachusetts, USA.
  • 4UNC Center for Bioethics, University of North Carolina at Chapel Hill, Chapel Hill, North Carolina, USA.
  • 5Center for Medical Ethics and Health Policy, Baylor College of Medicine, Houston, Texas, USA.
  • 6Department of Clinical Investigation, Boston Children's Hospital, Boston, Massachusetts, USA.
  • 7Department of Medical Ethics and Health Policy, University of Pennsylvania Perelman School of Medicine, Philadelphia, Pennsylvania, USA.
  • 81] Department of Pediatrics, Harvard Medical School, Boston, Massachusetts, USA [2] Children's Hospital Informatics Program, Boston Children's Hospital, Boston, Massachusetts, USA.

Abstract

Purpose:Approaches to return individual results to participants in genomic research variably focus on actionability, duty to share, or participants' preferences. Our group at Boston Children's Hospital has prioritized participants' preferences by implementing the Gene Partnership, a genomic research repository, based on the "Informed Cohort" model that offers return of results in accordance with participant preferences. Recognizing that ethical oversight is essential, the Gene Partnership Informed Cohort Oversight Board was convened in 2009.Methods:Over 3 years, the Informed Cohort Oversight Board developed guidelines for the return of individual genomic research results.Results:The Informed Cohort Oversight Board defined its guiding principles as follows: to respect the developing autonomy of pediatric participants and parental decision-making authority by returning results consistent with participants' preferences and to protect participants from harm. Potential harms and strategies to eliminate harm were identified. Guidelines were developed for participant preferences that consider the child's development and family dynamics. The Informed Cohort Oversight Board agreed that to prevent harm, including harms related to interfering with a child's future autonomy, there will be results that should not be returned regardless of participant preferences.Conclusion:The Informed Cohort Oversight Board developed guidelines for the return of results that respect the preferences of parents, children, and adult participants while seeking to protect against harm.Genet Med 16 7, 547-552.

PMID:
24406460
[PubMed - in process]
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