Professional consensus has traditionally discouraged predictive genetic testing when no childhood interventions can reduce future morbidity or mortality. However, advances in genome sequencing and accumulating evidence that children and families cope adequately with predictive genetic information have weakened this consensus. The primary argument remaining against testing appeals to children’s “right to an open future.” It claims that the autonomy of the future adult is violated when others make an irreversible choice to obtain or disclose predictive genetic information during childhood. We evaluate this argument and conclude that children’s interest in an open future should not be understood as a right. Rather an open future is one significant interest to weigh against other important interests when evaluating decisions. Thus, predictive genetic testing is ethically permissible in principle, as long as the interests promoted outweigh potential harms. We conclude by offering an expanded model of children’s interests that might be considered in such circumstances, and present two case analyses to illustrate how this framework better guides decisions about predictive genetic testing in pediatrics.
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Institute of Medicine. Assessing genetic risks: implications for health and social policy. Washington, DC: The National Academies Press; 1994.
Clarke A, Working Party of the Clinical Genetics Society (UK). The genetic testing of children. J Med Genet. 1994;31:785–797.
American Society of Human Genetics Board of Directors, American College of Medical Genetics Board of Directors. Points to consider: ethical, legal, and psychosocial implications of genetic testing in children and adolescents. Am J Hum Genet. 1995;57:1233–1241.
American Academy of Pediatrics Committee on Bioethics. Ethical issues with genetic testing in pediatrics. Pediatrics. 2001;107:1451–1455.
Arbour L, Canadian Paediatric Society, Bioethics Committee. Guidelines for genetic testing of healthy children. Paediatr Child Health. 2003;8:42–45.
European Society of Human Genetics. Genetic testing in asymptomatic minors: recommendations of the European Society of Human Genetics. Eur J Hum Genet. 2009;17:720–721.
National Society of Genetic Counselors. NSGC position statement: genetic testing of minors for adult-onset conditions. 2017. https://www.nsgc.org/p/bl/et/blogaid=860. Accessed 20 January 2019.
Human Genetics Society of Australasia. Presymptomatic and predictive testing for children and young adults. 2014. https://www.hgsa.org.au/documents/item/272. Accessed 20 January 2019.
Wade CH, Wilfond BS, McBride CM. Effects of genetic risk information on children’s psychosocial wellbeing: a systematic review of the literature. Genet Med. 2010;12:317–326.
Wakefield CE, Hanlon LV, Tucker KM, et al. The psychological impact of genetic information on children: a systematic review. Genet Med. 2016;18:755–762.
Fryer A. Inappropriate genetic testing of children. Arch Dis Child. 2000;83:283–285.
Duncan R, Savulescu J, Gillam L, et al. An international survey of predictive genetic testing in children for adult onset conditions. Genet Med. 2005;7:390–396.
Mand C, Gillam L, Delatycki MB, Duncan RE. Predictive genetic testing in minors for late-onset conditions: a chronological and analytical review of the ethical arguments. J Med Ethics. 2012;38:519–524.
Fenwick A, Plantinga M, Dheensa S, Lucassen A. Predictive genetic testing of children for adult-onset conditions: negotiating requests with parents. J Genet Couns. 2017;26:244–250.
McCullough LB, Brothers KB, Chung WK, et al. Professionally responsible disclosure of genomic sequencing results in pediatric practice. Pediatrics. 2015;136:e974–982.
Wisconsin v. Yoder, et al. 406 U.S. 205 (1972).
Feinberg J. The child’s right to an open future. In: Feinberg J, ed. Freedom and fulfillment: philosophical essays. Princeton, NJ: Princeton University Press; 1992. p. 76–97.
Davis DS. Genetic dilemmas and the child’s right to an open future. Hastings Cent Rep. 1997;27:7–15.
Davis DS. Genetic dilemmas and the child’s right to an open future. Rutgers Law J. 1997;28:549–592.
Davis DS. Genetic dilemmas: reproductive technology, parental choices, and children’s futures. 2nd ed. New York: Oxford University Press; 2010.
Gooding HC, Wilfond B, Boehm K, Biesecker BB. Unintended messages: the ethics of teaching genetic dilemmas. Hastings Cent Rep. 2002;32:37–39.
Hercher L, Uhlmann WR, Hoffman EP, Gustafson S, Chen KM, Public Policy Committee of NSGC. Prenatal testing for adult-onset conditions: the position of the National Society of Genetic Counselors. J Gen Couns. 2016;25:1139–1145.
Mameli M. Reproductive cloning, genetic engineering and the autonomy of the child: the moral agent and the open future. J Med Ethics. 2007;33:87–93.
Davis DS. The parental investment factor and the child’s right to an open future. Hastings Cent Rep. 2009;39:24–27.
de Souza M. Regulating preimplantation genetic diagnosis in Australia: disability and parental choice. J Law Med. 2015;22:915–933.
Wilfond BS, Miller PS, Korfiatis C, et al. Navigating growth attenuation in children with profound disabilities: children’s interests, family decision-making, and community concerns. Hastings Cent Rep. 2010;40:27–40.
Bredenoord AL, Dondorp W, Pennings G, De Wert G. Ethics of modifying the mitochondrial genome. J Med Ethics. 2011;37:97–100.
Darby RJ. The child’s right to an open future: is the principle applicable to non-therapeutic circumcision? J Med Ethics. 2013;39:463–468.
Hainz T. The enhancement of children versus circumcision: a case of double moral standards? Bioethics. 2015;29:507–515.
Camporesi S. Bend it like Beckham! The ethics of genetically testing children for athletic potential. Sports Ethics Philos. 2013;7:175–185.
Loland S. Against genetic tests for athletic talent: the primacy of the phenotype. Sports Med. 2015;45:1229–1233.
Camporesi S, McNamee MJ. Ethics, genetic testing, and athletic talent: children’s best interests, and the right to an open (athletic) future. Physiol Genomics. 2016;48:191–195.
Graf WD, Nagel SK, Epstein LG, Miller G, Nass R, Larriviere D. Pediatric neuroenhancement: ethical, legal, social, and neurodevelopmental implications. Neurology. 2013;80:1251–1260.
Krutzinna JI. Beyond an open future. Camb Q Healthc Ethics. 2017;26:313–325.
Yu JH, Jamal SM, Tabor HK, Bamshad MJ. Self-guided management of exome and whole-genome sequencing results: changing the results return model. Genet Med. 2013;15:684–690.
Bredenoord AL, de Vries MC, van Delden JJ. Next-generation sequencing: does the next generation still have a right to an open future? Nat Rev Genet. 2013;14:306.
Bredenoord AL, de Vries MC, van Delden H. The right to an open future concerning genetic information. Am J Bioeth. 2014;14:21–23.
Hofmann B. Incidental findings of uncertain significance: to know or not to know—that is not the question. BMC Med Ethics. 2016;17:13.
Sundby A, Boolsen MW, Burgdorf KS, et al. Attitudes of stakeholders in psychiatry towards the inclusion of children in genomic research. Hum Genomics. 2018;12:12.
Kon AA. Ethical issues in decision-making for infants with disorders of sex development. Horm Metab Res. 2015;47:340–343.
Cutas D, Hens K. Preserving children’s fertility: two tales about children’s right to an open future and the margins of parental obligations. Med Health Care Philos. 2015;18:253–260.
Kranendork EJ, Ploem MC, Hennekam RC. Regulating biobanking with children’s tissue: a legal analysis and the experts’ view. Eur J Hum Genet. 2016;24:30–36.
Mason PH. Personal genomic testing, genetic inheritance, and uncertainty. J Bioeth Inq. 2017;14:583–584.
Morissey C, Walker RL. The ethics of general population preventive genomic sequencing: rights and social justice. J Med Philos. 2018;43:22–43.
Mintz RL, Loike JD, Fischbach RL Will CRISPR germline engineering close the door to an open future. Sci Eng Ethics 2018. https://doi.org/10.1007/s11948-018-0069-6. pp. 1–15. [Epub ahead of print].
Sziron M, Hildt E. Digital media, the right to an open future, and children 0-5. Front Psychol. 2018;9:2137.
Bloch M, Hayden MR. Opinion: predictive testing for Huntington disease in childhood: challenges and implications. Am J Hum Genet. 1990;46:1–4.
Harper PS, Clarke A. Should we test children for ‘adult’ genetic diseases? Lancet. 1990;335:1205–1206.
Borry P, Stultiens L, Nys H, et al. Presymptomatic and predictive genetic testing in minors: a systematic review of guidelines and position papers. Clin Genet. 2006;70:374–381.
Duncan RE. Predictive genetic testing in young people: when is it appropriate? J Paediatr Child Health. 2004;40:593–595.
Duncan RE, Delatycki MB. Predictive genetic testing in young people for adult-onset conditions: where is the empirical evidence? Clin Genet. 2006;69:8–16.
Borry P, Goffin T, Nys H, Dierickx K. Predictive genetic testing in minors for adult‐onset genetic diseases. Mt Sinai J Med. 2008;75:287–296.
Wilfond B, Ross LF. From genetics to genomics: ethics, policy, and parental decision-making. J Pediatr Psychol. 2009;34:639–647.
Parker M. Genetic testing in children and young people. Fam Cancer. 2010;9:15–18.
Clarke A. What is at stake in the predictive genetic testing of children? Fam Cancer. 2010;9:19–22.
Fenwick J. Are guidelines for genetic testing of children necessary? Fam Cancer. 2010;9:23–25.
Gilbar R. Genetic testing of children for familial cancers: a comparative legal perspective on consent, communication of information and confidentiality. Fam Cancer. 2010;9:75–87.
Anderson JA, Hayeems RZ, Shuman C, et al. Predictive genetic testing for adult-onset disorders in minors: a critical analysis of the arguments for and against the 2013 ACMG guidelines. Clin Genet. 2015;87:301–310.
Mills C. The child’s right to an open future? J Soc Philos. 2003;34:499–509.
Cohen CB. Wrestling with the future: should we test children for adult-onset genetic conditions? Kennedy Inst Ethics J. 1998;8:111–130.
Millum J. The foundation of the child’s right to an open future. J Soc Philos. 2014;45:522–538.
Ross LF. Children, families, and health care decision making. New York: Clarendon Press; 1998.
Harris J, Keywood K. Ignorance, information and autonomy. Theor Med Bioeth. 2001;22:415–436.
Malpas P. Predictive genetic testing on children and respect for autonomy. Int J Child Right. 2005;13:273–285.
Robertson S, Savulescu J. Is there a case in favour of predictive genetic testing in young children? Bioethics. 2001;15:26–49.
Rhodes R. Why test children for adult-onset genetic diseases? Mt Sinai J Med. 2006;73:609–616.
Häyry M, Takala T. Genetic information, rights, and autonomy. Theor Med Bioeth. 2001;22:403–414.
Wenar L. Rights. Stanford Encyclopedia of Philosophy is Edward N. Zalta. Stanford, California: Center for the Study of Language and Information (CSLI), Stanford University; 2015.
British Society of Human Genetics (BSHG). Report on the genetic testing of children. 2010. https://www.bsgm.org.uk/media/678741/gtoc_booklet_final_new.pdf. Accessed 20 January 2019.
American Society of Human Genetics Board of Directors, American College of Medical Genetics Board of Directors. Points to consider: ethical, legal, and psychosocial implications of genetic testing in children and adolescents. Am J Hum Genet. 2015;97:6–21.
Ross LF. Theory and practice of pediatric bioethics. Perspect Biol Med. 2016;58:267–280.
Diekema DS. Parental refusals of medical treatment: the harm principle as threshold for state intervention. Theor Med Bioeth. 2004;25:243–264.
Gillam L. Children’s bioethics and the zone of parental discretion. Monash Bioeth Rev. 2010;20:01–03.
Malek J. What really is in a child’s best interest? Toward a more precise picture of the interests of children. J Clin Ethics. 2009;20:175–182.
Clayton EW. Genetic testing in children. J Med Philos. 1997;22:233–251.
Lucassen A, Fenwick A. Testing children for adult onset conditions: the importance of contextual clinical judgment. J Med Ethics. 2012;38:531–532.
Tan N, Amendola LM, O’Daniel JM, et al. Is “incidental finding” the best term?: a study of patients’ preferences. Genet Med. 2017;19:176–181.
Dworkin R. Rights as trumps. In: Waldron J, ed. Theories of rights. Oxford: Oxford University Press; 1984. p. 153–167.
Wilfond BS, Fernandez CV, Green RC. Disclosing secondary findings from pediatric sequencing to families: considering the “benefit to families”. J Law Med Ethics. 2015;43:552–558.
Creighton S, Almqvist EW, MacGregor D, et al. Predictive, pre-natal and diagnostic genetic testing for Huntington’s disease: the experience in Canada from 1987 to 2000. Clin Genet. 2003;63:462–475.
Morrison PJ, Harding-Lester S, Bradley A. Uptake of Huntington disease predictive testing in a complete population. Clin Genet. 2011;80:281–286.
Wilfond BS, Diekema DS. Engaging children in genomics research: decoding the meaning of assent in research. Genet Med. 2012;14:437–443.
Brothers KB, Lynch JA, Aufox SA, et al. Practical guidance on informed consent for pediatric participants in a biorepository. Mayo Clin Proc. 2014;89:1471–1480.
AAP Committee on Bioethics, AAP Committee on Genetics, ACMG Social Ethical and Legal Issues Committee. Ethical and policy issues in genetic testing and screening of children. Pediatrics. 2013;131:620–622.
The CSER Consortium is funded by National Human Genome Research Institute (NHGRI) and National Cancer Institute (NCI) (U01 HG006485 [Baylor College of Medicine], U01 HG006500 [Brigham and Women’s Hospital], U01 HG006546 [Children’s Hospital of Philadelphia], U01 HG006492 [Dana-Farber Cancer Institute], UM1 HG007301 [HudsonAlpha Institute], UM1 HG007292 [Kaiser Permanente], UM1 HG006508 [University of Michigan], U01 HG006487 [University of North Carolina], U01 HG006507 [University of Washington], R01 HG006615 [Boston Children’s Hospital], R21 HG006596 [Columbia University], R01 HG006600 [Columbia University], R21 HG006613 [Children’s Mercy Hospital], R21 HG006594 [Johns Hopkins University], R01 HG004500 [Mayo Clinic], R01 HG006618 [Seattle Children’s Hospital], R01 CA154517 [UC–San Francisco, Mayo College of Medicine, and University of Minnesota], R21 HG006612 [Vanderbilt University and McGill University], U01 HG007307 [University of Washington serving as the Coordinating Center]). ClinSeq (ZIA HG200387) is supported by the NHGRI Intramural Research Program. The authors thank the coordinating center of the Clinical Sequencing Exploratory Research (CSER) Consortium (University of Washington) for their support, and in particular Jeffrey Ou, who provided substantive support for this effort. Lucia Hindorff with NHGRI provided program staff support for this project. The authors also thank the following members of the CSER Consortium Pediatrics Working Group for participating in meetings to discuss the article as it was conceptualized and written: Benjamin Berkman (National Institutes of Health, Department of Bioethics), Barbara Bernhardt (University of Pennsylvania), Charlisse Caga-Anan (NCI), Ellen Wright Clayton (Vanderbilt University Medical Center), Aaron Goldenberg (Case Western Reserve University), Sara Chandros Hull (National Institutes of Health, Department of Bioethics), Steve Joffe (University of Pennsylvania), Ian Krantz (Children’s Hospital of Philadelphia), Michelle Lewis (John Hopkins Berman Institute of Bioethics), Wayne Liang (University of Alabama at Birmingham), Nicole Lockhart (NHGRI), Susana McCollum, Larry McCullough (Baylor College of Medicine), Amy McGuire (Baylor College of Medicine), Ali Noorbaksh (Baylor College of Medicine), Sarita Panchang (Baylor College of Medicine), D. Will Parsons (Baylor College of Medicine), Jacob Reiss (Kaiser Permanente Northwest), Myra Roche (University of North Carolina at Chapel Hill), Laura Rodriguez (NHGRI), Edward Romasko (Children’s Hospital of Philadelphia), Lainie Friedman Ross (University of Chicago), Richard Sharp (Mayo Clinic), Debra Skinner (University of North Carolina at Chapel Hill), Melody Slashinski (University of Massachusetts Amherst), Holly Tabor (Stanford University), Ashley Tomlinson (University of Pennsylvania), Susan Wolf (University of Minnesota), and Joon-Ho Yu (University of Washington). The authors also thank Leslie Ann McNolty (Center for Practical Bioethics) for helpful suggestions on revisions at several stages of the paper’s development, including the final draft. We would also like to thank Dena Davis (Lehigh University) who graciously served as a guest discussant for one of these meetings.
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