Parents, their children, whole exome sequencing and unsolicited findings: growing towards the child's future autonomy

A. Tibben*, W. Dondorp, C. Cornelis, N. Knoers, E. Brilstra, M. van Summeren, I. Bolt

*Corresponding author for this work

Research output: Contribution to journalArticleAcademicpeer-review

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Abstract

In a previous study we found that parents of children with developmental delay (DD) favoured acceptance of unsolicited findings (UFs) for medically actionable conditions in childhood, but that preferences diverged for UFs with no medical actionability, or only in adulthood, and regarding carrier status. Sometimes the child's future autonomy formed a reason for withholding UFs for the present, despite an unfavourable prognosis concerning the child's cognitive capabilities. This might be different for children undergoing whole exome sequencing (WES) for reasons other than DD and who are expected to exert future autonomy. This is the focus of the current study. We conducted nine qualitative, semi-structured interviews with parents of children, ages <1-15, after consenting to WES, but prior to feedback of results, and with three adolescent children. Several parents wished to receive any information that might in whatever way be relevant to the health and well-being of their child, and to a lesser extent wished the inclusion of information about non-actionable disorders and information concerning carrier status of autosomal recessive disorders. Although parents understood the rationale behind the centre's UFs disclosure policy, they also felt that they needed this information in order to be able to exert their parental responsibility and take good care of a child still dependent on them. Parents reason from their notion of parental responsibility but are also inclined to take adolescent children's preferences seriously and acknowledge the child's incipient autonomy as a ground for granting an increasing degree of self-determination on the road to adulthood.
Original languageEnglish
Pages (from-to)911-919
Number of pages9
JournalEuropean Journal of Human Genetics
Volume29
Issue number6
Early online date17 Jan 2021
DOIs
Publication statusPublished - Jun 2021

Keywords

  • communication
  • disclosure
  • ethics
  • incidental findings
  • patients views
  • perspectives
  • preferences
  • research participants
  • return
  • secondary variants
  • PATIENTS VIEWS
  • SECONDARY VARIANTS
  • INCIDENTAL FINDINGS
  • RESEARCH PARTICIPANTS
  • RETURN
  • DISCLOSURE
  • PREFERENCES
  • PERSPECTIVES
  • COMMUNICATION
  • ETHICS

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