Points to Consider: Ethical, Legal, and Psychosocial Implications of Genetic Testing in Children and Adolescents

Botkin, Jeffrey R.; Belmont, John W.; Berg, Jonathan S.; Berkman, Benjamin E.; Bombard, Yvonne; Holm, Ingrid A.; Levy, Howard P.; Ormond, Kelly E.; Saal, Howard M.; Spinner, Nancy B.; Wilfond, Benjamin S.; McInerney, Joseph D.

doi:10.1016/j.ajhg.2015.05.022

Cited by 470 publications

(363 citation statements)

References 95 publications

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“…Similarly, the risks of SV disclosure reported by our parental cohort have been identified before: potential insurance implications,4 10 15 33 34 psychological distress10 16 18 34 and inflicted insight,16 18 among others. However, we also uncovered novel forms of ambivalence toward SVs felt by parents of children undergoing WGS that have not been previously explored in this context.…”

Section: Discussionsupporting

confidence: 71%

Parents perspectives on whole genome sequencing for their children: qualified enthusiasm?

et al. 2016

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Despite general enthusiasm for WGS as a diagnostic test, many parents felt a duty to learn adult-onset SVs. Analogous to 'inflicted insight', we call this phenomenon 'inflicted ought'. Importantly, not all parents of children undergoing WGS view the best interests of their child in relational terms, thereby challenging an underlying justification for current ACMG guidelines for reporting incidental secondary findings from whole exome and WGS.

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Section: Discussionsupporting

confidence: 71%

Parents perspectives on whole genome sequencing for their children: qualified enthusiasm?

et al. 2016

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“…Recently, geneticists suggested asking for a separate consent for storage and uses beyond the screening program [36]; governance structures could be adapted to biobank standards. Further research is needed into these dynamics to find a balance between safeguarding the integrity of the screening program, on the one hand, and enabling secondary uses of blood spots with broad parental support and engagement, on the other hand.…”

Section: Discussionmentioning

confidence: 99%

Mothers' Views on Longer Storage of Neonatal Dried Blood Spots for Specific Secondary Uses

Teeffelen

Douglas

et al. 2015

Public Health Genomics

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Background: Neonatal dried blood spots (DBS) present a wealth of data. Currently, many countries discuss DBS storage, management and use. We collected data in the Netherlands on the awareness and views of an unheard stakeholder: mothers (-to-be). Methods: A survey was conducted on an Amsterdam pregnancy fair website in 2011. We included 1,272 women who were pregnant and/or had at least one child ≤5 years old. A descriptive analysis was used to score the awareness of and views on policies regarding the length of storage and secondary use. Results: 18.9% of mothers (n = 1,272) were aware of all five current DBS screening, storage and use policies. The views were positive for all ten potential specific secondary uses. Most support was given to etiology research, while test development by a company was least supported. Extending the DBS storage beyond the 5-year status quo was approved by 67.8% of the respondents, indefinite storage was approved by 54.7%. Conclusion: Mothers indicate support of several secondary uses and prolonged storage of DBS but report a low awareness of current storage and secondary use policies. Efforts must be made to involve parents as key stakeholders in DBS policies. This could be achieved through a parent-led advisory structure.

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“…How these challenges manifest psychosocially for the child and their future health behavior is also unknown. In general, current recommendations do not support genetic testing for adult onset disorders unless there are unusual circumstances, and do support allowing the child to reach the age of maturity to make their own decisions about testing (Botkin et al 2015). The qualitative results of this play illustrate that these recommendations remain controversial.…”

Section: Discussionmentioning

confidence: 99%

An empirical assessment of the short-term impacts of a reading of Deborah Zoe Laufer's drama Informed Consent on attitudes and intentions to participate in genetic research

Rothwell

Botkin

Cheek-O'Donnell

et al. 2018

AJOB Empirical Bioethics

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Objective: This study assessed the short-term impact of the play “Informed Consent” by Deborah Zoe Laufer (a fictionalized look at the controversy over specimens collected from the Havasupai Tribe for diabetes research in 1989) on perceptions of trust, willingness to donate biospecimens, attitudes toward harm and privacy among the medical and undergraduate students, faculty and the public in the intermountain west. Methods: Surveys were administered before and after a staged reading of the play by professional actors. Survey items included the short form Trust in Medical Researchers, and single item questions about group identity, ethics of genetic testing in children, and willingness to donate biospecimens. In addition, respondents were given the option to answer open-ended questions through email. Results: Out of the 481 who attended the play, 421 completed both the pre and post surveys, and 166 participants completed open-ended questions online approximately one week after the play. Across all participants, there were significant declines for Trust in Medical Researchers and for the survey item “is it ethical for investigators to test children for adult onset diseases,” (p < 0.001 for both) following the play. There was a significant increase in agreement to improve group identity protections (p < 0.001) and no differences on willingness to donate biospecimens to research (p = 0.777). Qualitative data provided extensive contextual data supporting these perspectives. Discussion: This is one of the first studies to document short-term impacts of a theatrical performance on both attitudes and behavioral intentions toward research ethics and clinical research participation. Future research should continue to explore the impact of theatrical performances among public and investigators on the ethical issues and complexities in clinical research.

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Points to Consider: Ethical, Legal, and Psychosocial Implications of Genetic Testing in Children and Adolescents

Cited by 470 publications

References 95 publications

Parents perspectives on whole genome sequencing for their children: qualified enthusiasm?

Parents perspectives on whole genome sequencing for their children: qualified enthusiasm?

Mothers' Views on Longer Storage of Neonatal Dried Blood Spots for Specific Secondary Uses

An empirical assessment of the short-term impacts of a reading of Deborah Zoe Laufer's drama Informed Consent on attitudes and intentions to participate in genetic research

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