title: Attitudes of parents toward the return of targeted and incidental genomic research findings in children.
abstract: OBJECTIVE
  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%).
  
  
  CONCLUSIONS
  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.
authors.affiliation: Department of Pediatrics, IWK Health Centre and Dalhousie University, Halifax, Nova Scotia, Canada.
authors.forename: Conrad V
authors.initials: CV
authors.lastname: Fernandez
authors.affiliation: Division of Pediatric Hematology/Oncology, Department of Pediatrics, The Hospital for Sick Children, University of Toronto, Toronto, Ontario, Canada.
authors.forename: Eric
authors.initials: E
authors.lastname: Bouffet
authors.affiliation: Division of Pediatric Hematology/Oncology, Department of Pediatrics, The Hospital for Sick Children, University of Toronto, Toronto, Ontario, Canada.
authors.forename: David
authors.initials: D
authors.lastname: Malkin
authors.affiliation: Department of Pediatrics, McGill University/McGill University Health Center, Montreal, Quebec, Canada.
authors.forename: Nada
authors.initials: N
authors.lastname: Jabado
authors.affiliation: Perinatal Epidemiology Research Unit, IWK Health Centre, Halifax, Nova Scotia, Canada.
authors.forename: Colleen
authors.initials: C
authors.lastname: O'Connell
authors.affiliation: Centre of Genomics and Policy, Department of Human Genetics, Faculty of Medicine, McGill University, Montreal, Quebec, Canada.
authors.forename: Denise
authors.initials: D
authors.lastname: Avard
authors.affiliation: Centre of Genomics and Policy, Department of Human Genetics, Faculty of Medicine, McGill University, Montreal, Quebec, Canada.
authors.forename: Bartha M
authors.initials: BM
authors.lastname: Knoppers
authors.affiliation: Maritime Medical Genetics Service, IWK Health Centre, Halifax, Nova Scotia, Canada.
authors.forename: Meghan
authors.initials: M
authors.lastname: Ferguson
authors.affiliation: Children's Hospital of Eastern Ontario Research Institute, University of Ottawa, Ottawa, Ontario, Canada.
authors.forename: Kym M
authors.initials: KM
authors.lastname: Boycott
authors.affiliation: 1] 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.
authors.forename: Poul H
authors.initials: PH
authors.lastname: Sorensen
authors.affiliation: Department of Ophthalmology and Visual Sciences, Dalhousie University, Halifax, Nova Scotia, Canada.
authors.forename: Andrew C
authors.initials: AC
authors.lastname: Orr
authors.affiliation: Department of Ophthalmology and Visual Sciences, Dalhousie University, Halifax, Nova Scotia, Canada.
authors.forename: Johane M
authors.initials: JM
authors.lastname: Robitaille
authors.affiliation: Department of Pharmacology, Dalhousie University, Halifax, Nova Scotia, Canada.
authors.forename: Christopher R
authors.initials: CR
authors.lastname: McMaster
mesh_terms: D000328:Adult; D002170:Canada; D002648:Child; D005260:Female; D036281:Genetic Research; D023281:Genomics; D007722:Health Knowledge, Attitudes, Practice; D006801:Humans; D033162:Incidental Findings; D008297:Male; D008875:Middle Aged; D010290:Parents / Q000523:psychology*; D011795:Surveys and Questionnaires