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Pediatric Research and the Return of Individual Research Results

Published online by Cambridge University Press:  01 January 2021

Denise Avard ,
Karine Sénécal ,
Parvaz Madadi  and
Daniel Sinnett
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Extract

As a matter of respect for the person, it is considered an ethical duty to offer to return research results to participants where appropriate. Nevertheless, the return of individual research results to participants raises many socio-ethical issues and greater challenges when the participant is a child. This discrepancy arises partly because the return of individual pediatric research results entails a tripartite relationship between researcher, child, and parent(s) and is embroiled in numerous considerations (e.g., acting in the best interest of the child, respect for the person, and respect for the autonomy of the parents/child).

Extra caution is required in the pediatric research context because children cannot generally decide (consent) whether they want to be informed of their own research results or whether the results should be disclosed to parents. Children have long been considered a special and vulnerable group, and their parents, as guardians, play a critical role in the consent process. However, with regards to the return of individual research results, this might pose a potential conflict of interest between the current or future desires of the child and those of the parents.

Type
Symposium
Information
Journal of Law, Medicine & Ethics , Volume 39 , Issue 4 , Winter 2011 , pp. 593 - 604
Copyright
Copyright © American Society of Law, Medicine and Ethics 2011

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References

Avard, D. Samuël, J. Knoppers, B. M., Paediatric Research in Canada (Montreal:, Éditions Thémis, 2009): at 23.Google Scholar
Id., at 24; Avard, D. Samuël, J. Black, L. Griener, G. Knoppers, B. M., Best Practices for Health Research Involving Children and Adolescents: Genetic, Pharmaceutical, Longitudinal Studies and Palliative Care Research, Center for Genomics and Policy, McGill University, 2011, at s. 8.Google Scholar
Patenaude, A. F., “Pediatric Psychology Training and Genetics: What Will Twenty-First-Century Pediatric Psychologists Need to Know?” Journal of Pediatric Psychology 28, no. 2 (2003): 135–145.CrossRefGoogle Scholar
Wilfond, B. Ross, L. F., “From Genetics to Genomics: Ethics, Policy, and Parental Decision-Making,” Journal of Pediatric Psychology 34, no. 6 (2009): 639–647.CrossRefGoogle Scholar
Rigby, H. Fernandez, C. V., “Providing Research Results to Study Participants: Support Versus Practice of Researchers Presenting at the American Society of Hematology Annual Meeting,” Blood 106, no. 4 (2005): 1199–1202, at 1201.CrossRefGoogle Scholar
Samuël, J. Knoppers, B. M. Avard, D., “Research Involving Children: An Analysis of International and Canadian Norms,” in Avard, et al, supra note 1, at Chap. 1, 1–32.Google Scholar
Hens, K. Nys, H. Cassiman, J. J. Dierickx, K., “Children, Biobanks and the Scope of Parental Consent,” European Journal of Human Genetics 19, no. 7 (2011): 735–739; Knoppers, B. M. Avard, D. Cardinal, G. Glass, K. C., “Children and Incompetent Adults in Genetic Research: Consent and Safeguards,” Nature Reviews Genetics 3, no. 3 (2002): 221–225.CrossRefGoogle Scholar
See Patenaude, , supra note 3.Google Scholar
Id.Google Scholar
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.CrossRefGoogle Scholar
Ormond, K. E. Wheeler, M. T. Hudgins, L. Klein, T. E. Butte, A. J. Altman, R. B. Ashley, E. A. Greely, H. T., “Challenges in the Clinical Application of Whole-Genome Sequencing,” The Lancet 375, no. 9727 (2010): 1749–1751.CrossRefGoogle Scholar
Kaye, J. Boddington, P. de Vries, J. Hawkins, N. Melham, K., “Ethical Implications of the Use of Whole Genome Methods in Medical Research,” European Journal of Human Genetics 18, no. 4 (2009): 398–403; Mayer, A. N. Dimmock, D. P. Arca, M. J. Bick, D. P. Verbsky, J. W. Worthey, E. A. Jacob, H. J. Margolis, D. A., “A Timely Arrival for Genomic Medicine,” Genetics in Medicine 13, no. 3 (2011): 195–196; 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.CrossRefGoogle Scholar
Ferri, E. Bynner, J. Wadsworth, M., Changing Britain, Changing Lives (London: Institute of Education Press, 2003).Google Scholar
See Avard, et al, supra note 2, s. 8.3.3.Google Scholar
Samuël, J. Ries, N. M. Malkin, D. Knoppers, B. M., “Biobanks and Children: Comparative International Policies,” in Avard, et al, at Chap. 4, 103–128, at 107; Samuël, J. Ries, N. M. Malkin, D. Knoppers, B. M., “Biobanks and Longitudinal Studies: Where Are the Children?” GenEdit 6, no. 3 (2008): 1–8.Google Scholar
Ries, N. LeGrandeur, J. Caulfield, T., “Handling Ethical, Legal and Social Issues in Birth Cohort Studies Involving Genetic Research: Responses from Studies in Six Countries,” BMC Medical Ethics 11, no. 1 (2010): 4–4.CrossRefGoogle Scholar
Madadi, P. Joly, Y. Avard, D. Chitayat, D. Smith, M. Ross, C. Carleton, B. Hayden, M. Koren, G., “Communicating Pharmacogenetic Research Results to Breastfeeding Mothers Taking Codeine: A Pilot Study of Perceptions and Benefits,” Clinical Pharmacology & Therapeutics 88, no. 6 (2010): 792–795.CrossRefGoogle Scholar
Partridge, A. H. Burstein, H. J. Gelman, R. S. Marcom, P. K. Winer, E. P., “Do Patients Participating in Clinical Trials Want to Know Study Results?” Journal of the National Cancer Institute 95, no. 6 (2003): 491–492.CrossRefGoogle Scholar
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, no. 11 (2008): 36–43.CrossRefGoogle Scholar
Centre of Genomics and Policy, Humgen, available at <http://www.humgen.org/int/search.cfm?mod=1&lang=1> (last visited September 8, 2011)+(last+visited+September+8,+2011)>Google Scholar
Ministre de la Santé et des Services Sociaux, Recueil De Textes Normatifs, available at <http://ethique.msss.gouv.qc.ca/site/fr_recueil.phtml> (last visited May 5, 2011).+(last+visited+May+5,+2011).>Google Scholar
U.S. Department of Health and Human Service, Office for Human Research Protections, International Compilation of Human Research Protections, Washington, D.C., 2011, available at <http://www.hhs.gov/ohrp/international/intlcompilation/hspcompilation-v20101130.pdf> (last visited August 16, 2011).+(last+visited+August+16,+2011).>Google Scholar
European Medicines Agency (EMEA), Ethical Considerations for Clinical Trials on Medicinal Products Conducted with the Paediatric Population (Recommendations of the Ad Hoc Group for the Development of Implementing Guidelines for Directive 2001/20/Ec Relating to Good Clinical Practice in the Conduct of Clinical Trials on Medicinal Products for Human Use, 2008, available at <http://ec.europa.eu/health/fles/paediatrics/docs/paeds_ethics_consultation20060929_en.pdf> (last visited April 7, 2011), s. 19.1.+(last+visited+April+7,+2011),+s.+19.1.>Google Scholar
Canadian Paediatric Society, “Ethical Issues in Health Research in Children,” Paediatric Child Health 13, no. 8 (2008): 707–712 [hereinafter cited as Ethical Issues in Health Research in Children]; Canadian Paediatric Society and Canadian College of Medical Geneticists, “Guidelines for Genetic Testing of Healthy Children - Addendum,” Paediatric Child Health 13, no. 4 (2008): 311–312; Réseau de Médecine Génétique Appliquée (RMGA), Statement of Principles for Genetic Research Involving Children, 2007), available at <http://www.rmga.qc.ca/fr/programs_and_forms> (last visited September 8, 2011).+(last+visited+September+8,+2011).>Google Scholar
Wilfond, B. S. Carpenter, K. J., “Incidental Findings in Pediatric Research,” Journal of Law, Medicine & Ethics 36, no. 2 (2008): 332–340.CrossRefGoogle Scholar
Id.Google Scholar
Bhatia, S., “Late Effects among Survivors of Leukemia during Childhood and Adolescence,” Blood Cells, Molecules, and Diseases 31, no. 1 (2003): 84–92; Oeffinger, K. C. Hudson, M. M., “Long Term Complications Following Childhood and Adolescent Cancer: Foundations for Providing Risk Based Health Care for Survivors,” CA: A Cancer Journal for Clinicians 54, no. 4 (2004): 208–236; Oeffinger, K. Mertens, A. Sklar, C. Kawashima, T. Hudson, M. Meadows, A. Friedman, D. et al, “Childhood Cancer Survivor Study (2006) Chronic Health Conditions in Adult Survivors of Childhood Cancer,” New England Journal of Medicine 355, no. 15 (2006): 1572–1582; Mody, R. Li, S. Dover, D. C. Sallan, S. Leisenring, W. Oeffinger, K. C. Yasui, Y. Robison, L. L. Neglia, J. P., “Twenty-Five-Year Follow-up among Survivors of Childhood Acute Lymphoblastic Leukemia: A Report from the Childhood Cancer Survivor Study,” Blood 111, no. 12 (2008): 5515–5523.CrossRefGoogle Scholar
Canadian Institutes of Health Research, Natural Sciences and Engineering Research Council of Canada and Social Sciences and Humanities Research Council of Canada, Tri-Council Policy Statement: Ethical Conduct for Research Involving Humans, 2010, at Commentary s. 13.2.Google Scholar
See Wilfond, Carpenter, , supra note 25.Google Scholar
Id.Google Scholar
United Nations, Convention on the Rights of the Child, Geneva, 1989, at s. 3.1, available at <http://www2.ohchr.org/english/law/crc.htm> (last visited August 16, 2011); Civil Code of Quebec. S.Q. 1991, c. 64. 1994, s. 33.+(last+visited+August+16,+2011);+Civil+Code+of+Quebec.+S.Q.+1991,+c.+64.+1994,+s.+33.>Google Scholar
Borry, P. Evers-Kiebooms, G. Cornel, M. C. Clarke, A. Dierickx, K., “Genetic Testing in Asymptomatic Minorsbackground Considerations Towards Eshg Recommendations,” European Journal of Human Genetics 17, no. 6 (2009): 711–719.CrossRefGoogle Scholar
World Health Organization (WHO), Genetic Databases: Assessing the Benefits and the Impact on Human & Patients Rights, Geneva, 2003, available at <http://www.who.int/genomics/publications/en/index5.html> (last visited September 8, 2011); United Nations Educational Scientific and Cultural Organization (UNESCO), International Declaration on Human Genetic Data, Paris, 2003, available at <http://portal.unesco.org/en/ev.php-URL_ID=17720&URL_DO=DO_TOPIC&URL_SECTION=201.html> (last visited August 16, 2011); Human Genome Organization (HUGO), Statement on the Principled Conduct of Genetics Research, Singapore, 1995, available at <http://www.hugo-international.org/img/statmentontheprincipledconductofgeneticsresearch.pdf> (last visited August 16, 2011); Organisation for Economic Co-operation and Development, Guidelines on Human Biobanks and Genetic Research Databases, 2009, at s. 4.14, available at <http://www.oecd.org/dataoecd/41/47/44054609.pdf> (last visited August 16, 2011); Council of Europe, “Additional Protocol to the Convention on Human Rights and Biomedicine, Concerning Biomedical Research,” 2005, at s. 27, available at <http://conventions.coe.int/Treaty/EN/Treaties/Html/195.htm> (last visited September 8, 2011); Canadian Institutes of Health Research, Natural Sciences and Engineering Research Council of Canada and Social Sciences and Humanities Research Council of Canada, Tri-Council Policy Statement: Ethical Conduct for Research Involving Humans, 2010, at s. 13.2–13.3, available at <http://www.pre.ethics.gc.ca/pdf/eng/tcps2/TCPS_2_FINAL_Web.pdf> (last visited September 8, 2011).+(last+visited+September+8,+2011);+United+Nations+Educational+Scientific+and+Cultural+Organization+(UNESCO),+International+Declaration+on+Human+Genetic+Data,+Paris,+2003,+available+at++(last+visited+August+16,+2011);+Human+Genome+Organization+(HUGO),+Statement+on+the+Principled+Conduct+of+Genetics+Research,+Singapore,+1995,+available+at++(last+visited+August+16,+2011);+Organisation+for+Economic+Co-operation+and+Development,+Guidelines+on+Human+Biobanks+and+Genetic+Research+Databases,+2009,+at+s.+4.14,+available+at++(last+visited+August+16,+2011);+Council+of+Europe,+“Additional+Protocol+to+the+Convention+on+Human+Rights+and+Biomedicine,+Concerning+Biomedical+Research,”+2005,+at+s.+27,+available+at++(last+visited+September+8,+2011);+Canadian+Institutes+of+Health+Research,+Natural+Sciences+and+Engineering+Research+Council+of+Canada+and+Social+Sciences+and+Humanities+Research+Council+of+Canada,+Tri-Council+Policy+Statement:+Ethical+Conduct+for+Research+Involving+Humans,+2010,+at+s.+13.2–13.3,+available+at++(last+visited+September+8,+2011).>Google Scholar
Patenaude, A. F. Basili, L. Fairclough, D. L. Li, F. P., “Attitudes of 47 Mothers of Pediatric Oncology Patients toward Genetic Testing for Cancer Predisposition,” Journal of Clinical Oncology 14, no. 2 (1996): 415–421.CrossRefGoogle Scholar
See Hens, et al, supra note 10.Google Scholar
See Avard, et al, supra note 2.Google Scholar
See Wilfond, Carpenter, , supra note 25.Google Scholar
See Hens, et al, supra note 10.Google Scholar
Elger, B. S. Harding, T. W., “Testing Adolescents for a Hereditary Breast Cancer Gene (BRCA1): Respecting Their Autonomy Is in Their Best Interest,” Archives of Pediatrics and Adolescent Medicine 154, no. 2 (2000): 113–119.CrossRefGoogle Scholar
Burke, W. Diekema, D. S., “Ethical Issues Arising from the Participation of Children in Genetic Research,” Journal of Pediatrics 149, no. 1 (2006): S34–S38; Borry, P. Evers-Kiebooms, G. Cornel, M. C. Clarke, A. Dierickx, K., “Genetic Testing in Asymptomatic Minorsbackground Considerations towards Eshg Recommendations,” European Journal of Human Genetics 17, no. 6 (2009): 711–719; Codori, A. M. Zawacki, K. L. Petersen, G. M. Miglioretti, D. L. Bacon, J. A. Trimbath, J. D. Booker, S. V. Picarello, K. Giardiello, F. M., “Genetic Testing for Hereditary Colorectal Cancer in Children: Long Term Psychological Effects,” American Journal of Medical Genetics Part A 116, no. 2 (2003): 117–128; see Ethical Issues in Health Research in Children, supra note 24.CrossRefGoogle Scholar
European Society of Human Genetics, Genetic Testing in Asymptomatic Minors, 2009, available at <https://www.eshg.org/fleadmin/eshg/documents/TestingInMinorsRecommendations.pdf> (last visited April 8, 2011); Borry, P. Stultiëns, L. Nys, H. Cassiman, J., “Presymptomatic and Predictive Genetic Testing in Minors: A Systematic Review of Guidelines and Position Papers,” Clinical Genetics 70, no. 5 (2006): 374–381.CrossRefGoogle Scholar
See Ethical Issues in Health Research in Children, supra note 24; Isasi, R. M. Knoppers, B. M., “Beyond the Permissibility of Embryonic and Stem Cell Research: Substantive Requirements and Procedural Safeguards,” Human Reproduction 21, no. 10 (2006): 2474–2481; Réseau de Médecine Génétique Appliquée (RMGA), Statement of Principles for Genetic Research Involving Children, 2007), available at <https://rmga.ca/files/attachments/0000/0057/FINAL_RMGA-Policy_Statement_on_Genetic_Research_with_Children.pdf> (last visited November 1, 2010).CrossRefGoogle Scholar
See Canadian Institutes of Health Research, supra note 28, at Commentary s. 13.2.Google Scholar
See Canadian Paediatric Society and Canadian College of Medical Geneticists, supra note 24.Google Scholar
Id.Google Scholar
Id.Google Scholar
See RMGA, supra note 42; Fabsitz, R. R. McGuire, A. Sharp, R. R. Puggal, M. Beskow, L. M. Biesecker, L. G. Bookman, E. et al, “Ethical and Practical Guidelines for Reporting Genetic Research Results to Study Participants,” Circulation: Cardiovascular Genetics 3, no. 6 (2010): 574–580, at 575.Google Scholar
See Burke, Diekema, , supra note 40.Google Scholar
European Society of Human Genetics, Genetic Testing in Asymptomatic Minors, 2009, available at <https://www.eshg.org/fleadmin/eshg/documents/TestingInMinorsRecommendations.pdf> (last visited August 15, 2011), at Recommendation 7; Canadian Paediatric Society and Canadian College of Medical Geneticists, “Guidelines for Genetic Testing of Healthy Children - Addendum,” Paediatric Child Health 13, no. 4 (2008): 311–312, at 45.+(last+visited+August+15,+2011),+at+Recommendation+7;+Canadian+Paediatric+Society+and+Canadian+College+of+Medical+Geneticists,+“Guidelines+for+Genetic+Testing+of+Healthy+Children+-+Addendum,”+Paediatric+Child+Health+13,+no.+4+(2008):+311–312,+at+45.>Google Scholar
Id. (European Society of Human Genetics).Google Scholar
Id., at Recommendations 10 and 11.Google Scholar
See Hens, et al, supra note 10.Google Scholar
Wilfond, B. Ross, L. F., “From Genetics to Genomics: Ethics, Policy, and Parental Decision-Making,” Journal of Pediatric Psychology 34, no. 6 (2009): 639–647, at 644.CrossRefGoogle Scholar
See Madadi, et al, supra note 17.Google Scholar
See Ethical Issues in Health Research in Children, supra note 24.Google Scholar
Forsberg, J. S. Hansson, M. G. Eriksson, S., “Changing Perspectives in Biobank Research: From Individual Rights to Concerns about Public Health Regarding the Return of Results,” European Journal of Human Genetics 17, no. 12 (2009): 1544–1549, at 1545; see Hens, et al, supra note 10.CrossRefGoogle Scholar
Réseau de Médecine Génétique Appliquée (RMGA), Statement of Principles for Genetic Research Involving Children, 2007, at 1, available at <https://rmga.ca/fles/attachments/0000/0057/FINAL_RMGA-Policy_Statement_on_Genetic_Research_with_Children.pdf> (last visited November 1, 2010).+(last+visited+November+1,+2010).>Google Scholar
Id., at 12.Google Scholar
Rosas-Blum, E. Shirsat, P. Leiner, M., “Communicating Genetic Information: A Difficult Challenge for Future Pediatricians,” BMC Medical Education 7, no. 1 (2007): 17–24.CrossRefGoogle Scholar
Baars, M. J. H. Henneman, L. ten Kate, L. P., “Deficiency of Knowledge of Genetics and Genetic Tests among General Practitioners, Gynecologists, and Pediatricians: A Global Problem,” Genetics in Medicine 7, no. 9 (2005): 605–610.CrossRefGoogle Scholar
See Canadian Paediatric Society, supra note 44.Google Scholar
Bookman, E. B. Langehorne, A. A. Eckfieldt, J. H. Glass, K. C. Jarvik, G. P. Klag, M. Koski, G. et al, “Reporting Genetic Results in Research Studies: Summary and Recommendations of an Nhlbi Working Group,” American Journal of Medical Genetics Part A 140, no. 10 (2006): 1033–1040.CrossRefGoogle Scholar
Meacham, M. C. Starks, H. Burke, W. Edwards, K., “Researcher Perspectives on Disclosure of Incidental Findings in Genetic Research,” Journal of Empirical Research on Human Research Ethics 5, no. 3 (2010): 31–41.CrossRefGoogle Scholar
See Fabsitz, et al, supra note 47, at 577; Canadian Paediatric Society and Canadian College of Medical Geneticists, supra note 44; Bookman, , supra note 62.Google Scholar
See Canadian Institutes of Health Research, supra note 28.Google Scholar
PHG Foundation, Public Health in an Era of Genome-Based and Personalised Medicine, Cambridge, 2010, available at <http://www.phgfoundation.org/file/6616/> (last visited August 15, 2011).+(last+visited+August+15,+2011).>Google Scholar
United Nations Educational Scientific and Cultural Organization (UNESCO), Report on Confidentiality and Genetic Data, Paris, 2000, at para. 51, available at <http://unesdoc.unesco.org/images/0013/001323/132334e.pdf> (last visited August 15, 2011).+(last+visited+August+15,+2011).>Google Scholar
Canadian Institutes of Health Research, supra note 28, at 181.Google Scholar
See Ries, LeGrandeur, Caulfield, , supra note 16.Google Scholar
See Ethical Issues in Health Research in Children, supra note 24.Google Scholar
Hunter, A. G. W. Sharpe, N. Mullen, M. Meschino, W. S., “Ethical, Legal, and Practical Concerns about Recontacting Patients to Inform Them of New Information: The Case in Medical Genetics,” American Journal of Medical Genetics 103, no. 4 (2001): 265–276.CrossRefGoogle Scholar
See Wilfond, Carpenter, , supra note 25, at 337.CrossRefGoogle Scholar
Fernandez, C. V. Santor, D. Weijer, C. Strahlendorf, C. Moghrabi, A. Pentz, R. Gao, J. Kodish, E., “The Return of Research Results to Participants: Pilot Questionnaire of Adolescents and Parents of Children with Cancer,” Pediatric Blood & Cancer 48, no. 4 (2007): 441–446.CrossRefGoogle Scholar
See Wilfond, Carpenter, , supra note 25.Google Scholar
Mayer, A. N. Dimmock, D. P. Arca, M. J. Bick, D. P. Verbsky, J. W. Worthey, E. A. Jacob, H. J. Margolis, D. A., “A Timely Arrival for Genomic Medicine,” Genetics in Medicine 13, no. 3 (2011): 195–196.CrossRefGoogle Scholar
See Fernandez, et al, supra note 73.Google Scholar