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‘We Should View Him as an Individual’: The Role of the Child’s Future Autonomy in Shared Decision-Making About Unsolicited Findings in Pediatric Exome Sequencing

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Abstract

In debates about genetic testing of children, as well as about disclosing unsolicited findings (UFs) of pediatric exome sequencing, respect for future autonomy should be regarded as a prima facie consideration for not taking steps that would entail denying the future adult the opportunity to decide for herself about what to know about her own genome. While the argument can be overridden when other, morally more weighty considerations are at stake, whether this is the case can only be determined in concrete cases. Importantly, when children grow into adolescents, respect for future autonomy will have to give way to respecting their emerging autonomy. When pediatric exome sequencing is done for complex conditions not involving developmental delay, respect for the child’s future or emerging autonomy should be a primary consideration for those charged with deciding on behalf of the child. Building on what Emanuel and Emanuel have termed the ‘deliberative model’ of shared decision making, we argue that if parents fail to give these considerations their due, professionals should actively invite them to do so. Taking a directive stance may be needed in order to make sure that the future or emerging autonomy of the child are duly considered in the decision-making process, but also to help the parents and themselves to shape their respective roles as responsible care-givers.

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Notes

  1. Findings unrelated to the reason for testing occur in all branches of medicine. They are often called ‘incidental findings’ (IFs). However, ‘incidental’ might suggest that such findings are rare, which is certainly not the case in clinical genomics. For this reason, the European Society of Human Genetics (ESHG) regards ‘unsolicited findings’ (UFs) as the more appropriate term in this context. [39] Conceptually, UFs (or IF)s are to be distinguished from so-called ‘Secondary findings’ (SFs). Although the latter also refer to results unrelated to the original reason for testing, SFs are actively sought for, whereas UFs are not. [30] It should be noted that both with regard to UFs and SFs, there is a risk of overdiagnosis, resulting from the fact that current predictivity data for the variants in question derive from evidence generated in affected families, whereas UFs or SFs are found in unaffected individuals [38].

  2. For an early dissenting voice see the contribution to this debate by Stephen Robertson and Julian Savulescu in their 2001 Bioethics paper [32].

  3. In their paper, Emanuel and Emanuel discuss two further models: the ‘informative’ and the ‘paternalistic’ model of the clinical relationship. In the former of these two, the professional limits herself to providing factual information, leaving it entirely to the patient to come to a decision; in the latter, the professional already knows what the best decision should be, and encourages the patient to consent to it. As there is no trace of shared decision making worth the name in either of these two models on the extremes of the 4-model spectrum, they are not relevant for our discussion.

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Funding

This research was funded by ZonMw—the Netherlands Organization for Health Research and Development (Grant No. 70-73000-98-047).

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Authors and Affiliations

  1. Department of Health, Ethics and Society, Faculty of Health, Medicine and Life Sciences, Maastricht University, De Byeplein 1, 6229 HA, Maastricht, The Netherlands

    W. Dondorp & G. De Wert

  2. Department of Medical Ethics, Philosophy and History of Medicine, Erasmus Medical Centre, PO Box 2040, 3000 CA, Rotterdam, The Netherlands

    I. Bolt

  3. Department of Clinical Genetics, Leiden University Medical Centre, Laan van Duivenvoorde 2-TO, 2252 AK, Voorschoten, The Netherlands

    A. Tibben

  4. Department of General Pediatrics, University Medical Centre Utrecht, UMC Utrecht, PO Box 85090, 3508 AB, Utrecht, The Netherlands

    M. Van Summeren

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  1. W. Dondorp
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All authors contributed to the design of the paper; a first draft was written by WD; IB, AT and GdW revised it for important intellectual content. All authors approved of the final version of the manuscript.

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Correspondence to W. Dondorp.

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Dondorp, W., Bolt, I., Tibben, A. et al. ‘We Should View Him as an Individual’: The Role of the Child’s Future Autonomy in Shared Decision-Making About Unsolicited Findings in Pediatric Exome Sequencing. Health Care Anal 29, 249–261 (2021). https://doi.org/10.1007/s10728-020-00425-7

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