title: Returning incidental findings from genetic research to children: views of parents of children affected by rare diseases.
abstract: OBJECTIVE
  To explore parental perceptions and experiences regarding the return of genomic incidental research findings in children with rare diseases.
  
  
  METHODS
  Parents of children affected by various rare diseases were invited to participate in focus groups or individual telephone interviews in Montreal and Ottawa. Fifteen participants were interviewed and transcriptions were analysed using thematic analysis.
  
  
  RESULTS
  Four emergent themes underscored parental enthusiasm for receiving incidental findings concerning their child's health: (1) right to information; (2) perceived benefits and risks; (3) communication practicalities: who, when, and how; and (4) service needs to promote the communication of incidental findings. Parents believed they should be made aware of all results pertaining to their child's health status, and that they are responsible for transmitting this information to their child, irrespective of disease severity. Despite potential negative consequences, respondents generally perceived a favourable risk-benefit ratio in receiving all incidental findings.
  
  
  CONCLUSIONS
  Understanding how parents assess the risks and benefits of returning incidental findings is essential to genomic research applications in paediatric medicine. The authors believe the study findings will contribute to establishing future best practices, although further research is needed to evaluate the impact of parental decisions on themselves and their child.
authors.affiliation: Centre of Genomics and Policy, McGill University, Montreal, Quebec, Canada.
authors.forename: Erika
authors.initials: E
authors.lastname: Kleiderman
authors.affiliation: Centre of Genomics and Policy, McGill University, Montreal, Quebec, Canada.
authors.forename: Bartha Maria
authors.initials: BM
authors.lastname: Knoppers
authors.affiliation: Centre of Genomics and Policy, McGill University, Montreal, Quebec, Canada.
authors.forename: Conrad V
authors.initials: CV
authors.lastname: Fernandez
authors.affiliation: Faculty of Medicine, Departments of Pediatrics and Bioethics, Dalhousie University, Halifax, Nova Scotia, Canada.
authors.forename: Kym M
authors.initials: KM
authors.lastname: Boycott
authors.affiliation: Children's Hospital of Eastern Ontario Research Institute, University of Ottawa, Ontario, Canada.
authors.forename: Gail
authors.initials: G
authors.lastname: Ouellette
authors.affiliation: Regroupement Québécois des Maladies Orphelines, Sherbrooke, Quebec, Canada.
authors.forename: Durhane
authors.initials: D
authors.lastname: Wong-Rieger
authors.affiliation: Institute for Optimizing Health Outcomes, Canadian Organization for Rare Disorders, Toronto, Ontario, Canada.
authors.forename: Shelin
authors.initials: S
authors.lastname: Adam
authors.affiliation: Department of Medical Genetics, University of British Columbia, Vancouver, British Columbia, Canada.
authors.forename: Julie
authors.initials: J
authors.lastname: Richer
authors.affiliation: Centre of Genomics and Policy, McGill University, Montreal, Quebec, Canada.
authors.forename: Denise
authors.initials: D
authors.lastname: Avard
mesh_terms: D000293:Adolescent; D000328:Adult; D002648:Child; D002675:Child, Preschool; D005260:Female; D036281:Genetic Research / Q000941:ethics*; D006801:Humans; D033162:Incidental Findings; D008297:Male; D008875:Middle Aged; D010290:Parents / Q000523:psychology*; D010372:Pediatrics / Q000941:ethics; D035583:Rare Diseases / Q000175:diagnosis / Q000523:psychology*; D055815:Young Adult