title: To disclose, or not to disclose? Context matters.
abstract: Progress in understanding childhood disease using next-generation sequencing (NGS) portends vast improvements in the nature and quality of patient care. However, ethical questions surrounding the disclosure of incidental findings (IFs) persist, as NGS and other novel genomic technologies become the preferred tool for clinical genetic testing. Thus, the need for comprehensive management plans and multidisciplinary discussion on the return of IFs in pediatric research has never been more immediate. The aim of this study is to explore the views of investigators concerning the return of IFs in the pediatric oncology research context. Our findings reveal at least four contextual themes underlying the ethics of when, and how, IFs could be disclosed to participants and their families: clinical significance of the result, respect for individual, scope of professional responsibilities, and implications for the healthcare/research system. Moreover, the study proposes two action items toward anticipatory governance of IF in genetic research with children. The need to recognize the multiplicity of contextual factors in determining IF disclosure practices, particularly as NGS increasingly becomes a centerpiece in genetic research broadly, is heightened when children are involved. Sober thought should be given to the possibility of discovering IF, and to proactive discussions about disclosure considering the realities of young participants, their families, and the investigators who recruit them.
authors.affiliation: Centre of Genomics and Policy, McGill University, Montreal, Quebec, Canada.
authors.forename: Vasiliki
authors.initials: V
authors.lastname: Rahimzadeh
authors.affiliation: Centre of Genomics and Policy, McGill University, Montreal, Quebec, Canada.
authors.forename: Denise
authors.initials: D
authors.lastname: Avard
authors.affiliation: Centre of Genomics and Policy, McGill University, Montreal, Quebec, Canada.
authors.forename: Karine
authors.initials: K
authors.lastname: Sénécal
authors.affiliation: Centre of Genomics and Policy, McGill University, Montreal, Quebec, Canada.
authors.forename: Bartha Maria
authors.initials: BM
authors.lastname: Knoppers
authors.affiliation: Department of Pediatrics, University of Montreal, Montreal, Quebec, Canada.
authors.forename: Daniel
authors.initials: D
authors.lastname: Sinnett
mesh_terms: D000328:Adult; D000368:Aged; D035843:Biomedical Research; D030881:Disclosure / Q000941:ethics*; D005260:Female; D005820:Genetic Testing / Q000941:ethics; D006801:Humans; D033162:Incidental Findings; D008297:Male; D008495:Medical Oncology; D008875:Middle Aged; D010372:Pediatrics; D036301:Qualitative Research; D012108:Research Personnel / Q000941:ethics; D055815:Young Adult