Genet Med. 2014 Jan 16. doi: 10.1038/gim.2013.201. [Epub ahead of print]

Attitudes of parents toward the return of targeted and incidental genomic research findings in children.

Fernandez CV1, Bouffet E2, Malkin D2, Jabado N3, O'Connell C4, Avard D5, Knoppers BM5, Ferguson M6, Boycott KM7, Sorensen PH8, Orr AC9, Robitaille JM9,McMaster CR10.

Author information

• 1Department of Pediatrics, IWK Health Centre and Dalhousie University, Halifax, Nova Scotia, Canada.
• 2Division of Pediatric Hematology/Oncology, Department of Pediatrics, The Hospital for Sick Children, University of Toronto, Toronto, Ontario, Canada.
• 3Department of Pediatrics, McGill University/McGill University Health Center, Montreal, Quebec, Canada.
• 4Perinatal Epidemiology Research Unit, IWK Health Centre, Halifax, Nova Scotia, Canada.
• 5Centre of Genomics and Policy, Department of Human Genetics, Faculty of Medicine, McGill University, Montreal, Quebec, Canada.
• 6Maritime Medical Genetics Service, IWK Health Centre, Halifax, Nova Scotia, Canada.
• 7Children's Hospital of Eastern Ontario Research Institute, University of Ottawa, Ottawa, Ontario, Canada.
• 81] Department of Molecular Oncology, BC Cancer Research Centre, University of British Columbia, Vancouver, British Columbia, Canada [2] Department of Pathology, University of British Columbia, Vancouver, British Columbia, Canada.
• 9Department of Ophthalmology and Visual Sciences, Dalhousie University, Halifax, Nova Scotia, Canada.
• 10Department of Pharmacology, Dalhousie University, Halifax, Nova Scotia, Canada.

Abstract

Purpose:We describe parental attitudes toward the return of targeted and incidental genomic research results in the setting of high-risk pediatric cancer and inherited childhood diseases.

Methods:A validated 36-item questionnaire was mailed to participants in three large-scale genome research consortia examining attitudes toward receipt of genomic research results and the influence of certainty, severity, and onset of the condition, in addition to responsibilities to extended family and provision of results even after death of the proband.

Results:Of the 563 participants who were sent questionnaires, 362 (64%) responded. Most of them stated a positive right to receive results related to the target condition (97%) or to incidental findings (86%); no difference was found in results between participants with cancer and those with orphan diseases. Furthermore, 92% indicated that genomic research for childhood-onset conditions should occur. The majority wanted incidental results predicting susceptibility even to untreatable fatal conditions (83%), to multiple conditions (87%), or to those with uncertain impact (70%). Most felt sibling genomic results showing serious conditions, whether treatable (93%) or not (88%), and/or results discovered after death of the proband should be shared with family (74%).

Conclusion:Many parents of children in pediatric genomic research indicated a strong desire to receive a broader range of results than is described in consensus recommendations. Clear delineation of what will be offered should be established at the time of consent.