A Framework for Analyzing the Ethics of Disclosing Genetic Research Findings

For a discussion of these ethical principles and their implications for disclosure obligations, see Bredenoord A. L. , “Disclosure of Individual Genetic Data to Research Participants: The Debate Reconsidered,” Trends in Genetics 27, no. 2 (2011): 41–47;.

Bredenoord A. L. Onland-Moret N. C. Van Delden J. J. , “Feedback of Individual Genetic Results to Research Participants: In Favor of a Qualified Disclosure Policy,” Human Mutation 32, no. 8 (2011): 861–867.

Kollek R. Petersen I. , “Disclosure of Individual Research Results in Clinico-Genomic Trials: Challenges, Classification and Criteria for Decision-Making,” Journal of Medical Ethics 37, no. 5 (2011): 271–275.

Dressler L. G. Smolek S. Ponsaran R. Markey J. M. Starks H. Gerson N. Lewis S. , “IRB Perspectives on the Return of Individual Results from Genomic Research,” Genetics in Medicine 14, no. 2 (2012): 215–222, at 220.

Green R. C. Berg J. S. Grody W. W. Kalia S. S. Korf B. R. Martin C. L. McGuire A. L. , “ACMG Recommendations for Reporting of Incidental Findings in Clinical Exome and Genome Sequencing,” Genetics in Medicine 15, no. 7 (2013): 565–574.

Burke W. Matheny Antommaria H. A. H. Bennett R. Botkin J. Wright Clayton E. Henderson G. E. Holm I. A. , “Recommendations for Returning Genomic Incidental Findings? We Need to Talk!” Genetics in Medicine 15, no. 11 (2013): 854–859.

Presidential Commission for the Study of Bioethical Issues, Anticipate and Communicate: Ethical Management of Incidental and Secondary Findings (Presidential Commission for the Study of Bioethical Issues, December 2013), available at <http://bioethics.gov/sites/default/files/FINALAnticipate-Communicate_PCSBI_0.pdf>(last visited April 10, 2014).

Id., at 28–29.

This distinction is frequently traced back to Gallie's discussion of “essentially contested concepts” and to Rawls' elaboration of related issues in A Theory of Justice. Gallie W. B. , “Essentially Contested Concepts,” Proceedings of the Aristotelian Society 56 (1956): 167–198.

Rawls J. , A Theory of Justice (Cambridge, MA: Harvard University Press): At Chap. 32.

A list of these journal articles is available from the authors on request.

Christenhusz G. M. Devriendt K. Dierickx K. , “Secondary Variants – in Defense of a More Fitting Term in the Incidental Findings Debate,” European Journal of Human Genetics 21, no. 12 (2013): 1331–1334.

Tabor H. K. Berkman B. E. Chandros Hull S. Bamshad M. J. , “Genomics Really Gets Personal: How Exome and Whole Genome Sequencing Challenge the Ethical Framework of Human Genetics Research,” American Journal of Medical Genetics Part A, 155 (2011): 2916–2924.

Wolf S. M. Lawrenz F. P. Nelson C. A. Kahn J. P. Cho M. K. Wright Clayton E. Fletcher J. G. , “Managing Incidental Findings in Human Subjects Research: Analysis and Recommendations,” Journal of Law, Medicine & Ethics 36, no. 2 (2008): 219–248.

For examples of uptake, see Richardson H. S. , “Incidental Findings and Ancillary-Care Obligations,” Journal of Law, Medicine & Ethics 36, no. 2 (2008): 256–270;.

Simon C. M. Williams J. K. Shinkunas L. Brandt D. Daack-Hirsch S. Driessnack M. , “Informed Consent and Genomic Incidental Findings: IRB Chair Perspectives,” Journal of Empirical Research on Human Research Ethics: An International Journal 6, no. 4 (2011): 53–67;.

Van Ness B. , “Genomic Research and Incidental Findings,” Journal of Law, Medicine & Ethics 36, no. 2 (2008): 292–297.

Wolf S. M. Crock B. N. Van Ness B. Lawrenz F. Kahn J. P. Beskow L. M. Cho M. K. , “Managing Incidental Findings and Research Results in Genomic Research Involving Biobanks and Archived Data Sets,” Genetics in Medicine 14 (2012): 361–384, at 364;.

Wolf S. M. , “Introduction: The Challenge of Incidental Findings,” Journal of Law, Medicine & Ethics 36, no. 2 (2008): 216–218;.

see Wolf , supra note 12;.

Richardson , supra note 12, at 258.

See also Parker L. S. , “Best Laid Plans for Offering Results Go Awry,” American Journal of Bioethics 6, no. 6 (2006):22–23. (“Restricted by the study's hypothesis or research question.”).

Knoppers B. M. Dam A. , “Return of Results: Towards a Lexicon?” Journal of Law, Medicine & Ethics 39, no. 4 (2011): 577–582.

Miller F. G. Mello M. M. Joffe S. , “Incidental Findings in Human Subjects Research: What Do Investigators Owe Research Participants?” Journal of Law, Medicine & Ethics 36, no. 2 (2008): 271–279.

Terry S. F. , “The Tension Between Policy and Practice in Returning Research Results and Incidental Findings in Genomic Biobank Research,” Minnesota Journal of Law, Science & Technology 13, (2012): 691–925, at 705.

Cho M. K. , “Understanding Incidental Findings in the Context of Genetics and Genomics,” Journal of Law, Medicine & Ethics 36, no. 2 (2008): 280–285, at 281.

See also Kohane I. S. Taylor P. L. , “Multidimensional Results Reporting to Participants in Genomic Studies: Getting It Right,” Science Translational Medicine 2, no. 37 (2010): 37cm19, at 2.

Hens K. Nys H. Cassiman J. J. Dierickx K. , “The Return of Individual Research Findings in Paediatric Genetic Research,” Journal of Medical Ethics 37, no. 3 (2011): 179–183;.

Knoppers B. M. Rioux A. Zawati M. H. , “Pediatric Research ‘Personalized’? International Perspectives on the Return of Results,” Personalized Medicine 10, no. 1 (2013): 89–95.

See Richardson , supra note 12, at 258.

Id.

See Wolf , supra note 12

Equivalent constraints are included in Wolf, supra note 13.

See Knoppers Dam , supra note 14, at 580;%%.%

Miller Mello Joffe , supra note 15;.

Knoppers Rioux Zawati , supra note 18.

See Knoppers Dam , supra note 14, at 580.

See Kohane Taylor , supra note 17, at 2.

See Wolf , supra note 13, at 216.

See Knoppers Dam , supra note 14, at 580.

See Wolf , supra note 12. Emphasis added.

Parker L. S. , “The Future of Incidental Findings: Should They Be Viewed as Benefits?” Journal of Law, Medicine & Ethics 36, no. 2 (2008): 341–351, at 341.

Id., at 342.

See Wolf , supra note 12;.

Wolf , supra note 13, at 216.

See Richardson , supra note 12, at 258.

Id.

Id.

Ravitsky V. Wilfond B. S. , “Disclosing Individual Genetic Results to Research Participants,” American Journal of Bioethics 6, no. 6 (2006): 8–17;.

see Wolf , supra note 13;.

Bookman E. B. Langehorne A. A. Eckfeldt J. H. Glass K. C. Jarvik G. P. Klag M. Koski G. , “Reporting Genetic Results in Research Studies: Summary and Recommendations of an NHLBI Working Group,” American Journal of Medical Genetics Part A, 140A (2006): 1033–1040;.

Knoppers B. M. Joly Y. Simard J. Durocher F. , “The Emergence of an Ethical Duty to Disclose Genetic Research Results: International Perspectives,” European Journal of Human Genetics 14, no. 11 (2006): 1170–1178;.

Miller F. A. Christensen R. Giacomini M. Robert J. S. , “Duty to Disclose What? Querying the Putative Obligation to Return Research Results to Participants,” Journal of Medical Ethics 34, no. 3 (2008): 210–213 (using the term “data veracity”);.

Holm I. A. Taylor P. L. , “The Informed Cohort Oversight Board: From Values to Architecture,” Minnesota Journal of Law, Science & Technology 13, no. 2 (2012): 669–925;.

Parker L. S. , “Returning Individual Research Results: What Role Should People's Preferences Play?” Minnesota Journal of Law, Science & Technology 13, no. 2 (2012): 449–925.

See Ravitsky Wilfond , supra note 34, at 10.

See Wolf , supra note 12, at 237.

See Bookman , supra note 34, at 1034.

Fernandez C. V. Weijer C. , “Obligations in Offering to Disclose Genetic Research Results,” American Journal of Bioethics 6, no. 6 (2006): 44–46.

See Bredenoord , supra note 1, at 45.

See Dressler , supra note 3, at 219.

See Ravitsky Wilfond , supra note 34, at 10;.

Miller F. G. Shalowitz D. I. , “Disclosing Individual Results of Clinical Research: Implications of Respect for Participants,” JAMA 294, no. 6 (2005): 737–740;.

Holm Taylor , supra note 34.

See Knoppers , supra note 34, at 1175.

See Fernandez Weijer , supra note 38, at 45;.

Dressler L. G. Juengst E. T. , “Thresholds and Boundaries in the Disclosure of Individual Genetic Research Results,” American Journal of Bioethics 6, no. 6 (2006): 18–20.

Fabsitz R. R. McGuire A. Sharp R. R. Puggal M. Beskow L. M. Biesecker L. G. Bookman E. , “Ethical and Practical Guidelines for Reporting Genetic Research Results to Study Participants Updated Guidelines from a National Heart, Lung, and Blood Institute Working Group,” Circulation: Cardiovascular Genetics 3, no. 6 (2010): 574–580, at 577.

See Cho , supra note 17, at 282.

See Knoppers Dam , supra note 14, at 579.

Klitzman R. , “Questions, Complexities, and Limitations in Disclosing Individual Genetic Results,” American Journal of Bioethics 6 (2006): 34–36.

See, e.g., Knoppers Dam , supra note 14, at 579;.

Ravitsky Wilfond , supra note 34;.

Bookman , supra note 34;.

Murphy J. Scott J. Kaufman D. Geller G. LeRoy L. Hudson K. , “Public Expectations for Return of Results from Large-Cohort Genetic Research,” American Journal of Bioethics 8 (2008): 36–43;.

Parker , supra note 34.

See Bookman , supra note 34, at 1034.

See Ravitsky Wilfond , supra note 34, at 11.

See Bookman , supra note 34, at 1034.

See also Ormond K. E. , “Disclosing Genetic Research Results: Examples from Practice,” American Journal of Bioethics 6, no. 6 (2006): 30–32.

See Murphy , supra note 48.

See Parker , supra note 34.

See Ormond , supra note 51, at 31.

See Wolf , supra note 12, at 231;.

Holm Taylor , supra note 34;.

Van Ness , supra note 12.

See Wolf , supra note 12, at 231.

See Cho , supra note 17, at 282.

See, e.g., Dressler Juengst , supra note 43;.

Bredenoord , supra note 1.

Beskow L. M. Burke W. , “Offering Individual Genetic Research Results: Context Matters,” Science Translational Medicine 2, no. 38 (2010): 38cm20.

See Knoppers Dam , supra note 14, at 580. Similarly, Matthew Gordon describes “individual findings that are medically useful in the sense that there is some action that can be taken to prevent or ameliorate the condition indicated by the finding:” Gordon M. P. , “A Legal Duty to Disclose Individual Research Findings to Research Subjects,” Food and Drug Law Journal 64, no. 1 (2009): 225–260.

See Bookman , supra note 34.

See Bredenoord Onland-Moret Van Delden , supra note 1, at 865.

Id.

See Ravitsky Wilfond , supra note 34, at 11 (emphasis added).

See Wolf , “Managing Incidental Findings in Human Subjects Research,” supra note 12.

Id.

See Fernandez Weijer , supra note 38;.

Lavieri R. R. Garner S. A. , “Ethical Considerations in the Communication of Unexpected Information with Clinical Implications,” American Journal of Bioethics 6, no. 6 (2006): 46–48.

Brief E. Mackie J. Illes J. , “Incidental Findings in Genetic Research: A Vexing Challenge for Community Consent,” Minnesota Journal of Law, Science & Technology 13, no. 2 (2012): 541–558.

McGeveran W. Fatehi L. McGarraugh P. , “Deidentification and Reidentification in Returning Individual Findings from Biobank and Secondary Research: Regulatory Challenges and Models for Management,” Minnesota Journal of Law, Science & Technology 13, no. 2 (2012): 485–539.

See Wolf , supra note 12, at 225.

Shalowitz D. I. Miller F. G. , “Communicating the Results of Clinical Research to Participants: Attitudes, Practices, and Future Directions,” PLoS Medicine 5, no. 5 (2008): 0714–0720.

See Kohane Taylor , supra note 17, at 2–3.

See Knoppers , supra note 34, at 1174–1175.

Knoppers B. M. Laberge C. , “Return of ‘Accurate’ and ‘Actionable’ Results: Yes!” American Journal of Bioethics 9, no. 6 (2009): 107–109.

See Kohane Taylor , supra note 17, at 2.

Pullman D. Hodgkinson K. , “Genetic Knowledge and Moral Responsibility: Ambiguity at the Interface of Genetic Research and Clinical Practice,” Clinical Genetics 69, no. 3 (2006): 199–203, at 202.

Id.

McGuire A. L. Caulfield T. Cho M. K. , “Research Ethics and the Challenge of Whole-Genome Sequencing,” Nature Reviews: Genetics 9, no. 2 (2008): 152–156, at 153.

Renegar G. , “Returning Genetic Research Results to Individuals: Points-to-Consider,” Bioethics 20, no. 1 (2006): 24–36.

See Kollek Petersen , supra note 2, at 272.

Westbrook M. J. , “Mapping the Incidentalome: Estimating Incidental Findings Generated through Clinical Pharmacogenomics Testing,” Genetics in Medicine 15, no. 5 (2013): 325–331.

See Renegar , supra note 79.

See Miller , supra note 34.

See Knoppers Dam , supra note 14, at 579.

Abdul-Karim R. Berkman B. E. Wendler D. Rid A. Khan J. Badgett T. Chandros Hull S. , “Disclosure of Incidental Findings From Next-Generation Sequencing in Pediatric Genomic Research,” Pediatrics 131, no. 3 (2013): 564–571, at 567–568.

See, e.g., Miller , supra note 34;.

O'Daniel J. Haga S. B. , “Public Perspectives on Returning Genetics and Genomics Research Results,” Public Health Genomics 14, no. 6 (2011): 346–355, at 353;.

Wolf , supra note 12.

Affleck P. , “Is It Ethical to Deny Genetic Research Participants Individualised Results?” Journal of Medical Ethics 35, no. 4 (2009): 209–213, at 210.

See Gordon , supra note 60, at 229.

See also Wolf , supra note 12, who advocate broadening “utility” to include “what a research participant would find useful, recognizing not only treatment utility but also health or reproductive information utility.”.

See Beskow Burke , supra note 59. A similar definition is adopted in Wolf , supra note 13, at 373.

See Ormond , supra note 51, at 30.

See Ravitsky Wilfond , supra note 34.

See also Shalowitz , supra note 41, at 739.

See Terry , supra note 16.

Matsui K. Lie R. K. Kita Y. Ueshima H. , “Ethics of Future Disclosure of Individual Risk Information in a Genetic Cohort Study: A Survey of Donor Preferences,” Journal of Epidemiology 18, no. 5 (2008): 217–224, at 221.

See Terry , supra note 16, at 713.

See Fabsitz , supra note 44, at 575–576.

See Wolf , supra note 13, at 373.

See Murphy , supra note 48.

Rothstein M. A. , “Tiered Disclosure Options Promote the Autonomy and Well-Being of Research Subjects,” American Journal of Bioethics 6, no. 6 (2006): 20–21, at 21.

See also Parker , supra note 34.

See Parker , supra note 34.

See Miller Mello Joffe , supra note 15.

See Wolf , supra note 12, at 233.

See also Wolf , supra note 13, at 371.

See Terry , supra note 16, at 706.

See Matsui , supra note 93, at 222.

See Kohane Taylor , supra note 17, at 2–3.

See discussion in Gliwa C. Berkman B. E. , “Do Researchers Have an Obligation to Actively Look for Genetic Incidental Findings?” American Journal of Bioethics 13, no. 3 (2013): 32–42, at 32.

See, e.g., Affleck , supra note 87: “For the purposes of this paper, a research ‘result’ is simply regarded as a new piece of information that may relate to a particular individual.”.

Notably, a threshold question in this regard is whether a given piece of information constitutes a “finding,” the answer to which is likely to vary across different contexts and perspectives. The information that would constitute a “finding” for a scientist conducting research would likely be quite different to the level of certainty required for a clinician treating a patient.

Parens E. Appelbaum P. Chung W. , “Incidental Findings in the Era of Whole Genome Sequencing?” Hastings Center Report 43, no. 4 (2013): 16–19, at 18.

Stedman's Medical Dictionary, s.v. “Clinical,” Lathrop Stedman T. , ed. (Baltimore: Williams & Wilkins, 1995).

See Beskow Burke , supra note 59.

See Ravitsky Wilfond , supra note 34.

See Brief Mackie Illes , supra note 68.

Wolf , “Managing Incidental Findings in Human Subjects Research,” supra note 12.

See Knoppers , supra note 34.

See Matsui , supra note 93.

See Fabsitz , supra note 44.

See Murphy , supra note 48, using the phrase “categories of action.”.

See Beskow Burke , supra note 59.

See Ormond , supra note 51.

Merriam-Webster, s.v. “Volition,” available at <http://www.merriam-webster.com/dictionary/volition>(last visited April 11, 2014).

See Parker , supra note 34.

See Green , supra note 4.