Citizens’ Values Regarding Research With Stored Samples From Newborn Screening in Canada

Bombard, Yvonne; Miller, Fiona A.; Hayeems, Robin Z.; Carroll, June C.; Avard, Denise; Wilson, Brenda J.; Little, Julian; Bytautas, Jessica P; Allanson, Judith; Axler, Renata; Giguère, Yves; Chakraborty, Pranesh

doi:10.1542/peds.2011-2572

Cited by 42 publications

(63 citation statements)

References 37 publications

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“…[22][23][24][25][26] These deliberative methods are increasingly used to elicit public values on genomics policy issues. 17,24,25,27 The focus of this paper is on the panel's review and deliberations of GEP for early stage breast cancer, and the general topic of personalized medicine. The panel was asked to deliberate on the ethical and societal questions raised by these technologies, based on a list of 'moral' questions developed by BjØrn Hofmann.…”

Section: Data Collection and Analysismentioning

confidence: 99%

Citizens' perspectives on personalized medicine: a qualitative public deliberation study

et al. 2013

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Our objective was to explore citizens' informed and reasoned values and expectations of personalized medicine, a timely yet novel genomics policy issue. A qualitative, public deliberation study was undertaken using a citizens' reference panel on health technologies, established to provide input to the health technology assessment process in Ontario, Canada. The citizens' panel consisted of five women and nine men, aged 18-71 years, with one member selected from each health authority region. There were shared expectations among the citizens' panel members for the potential of personalized medicine technologies to improve care, provided they are deemed clinically valid and effective. These expectations were tempered by concerns about value for money and the possibility that access to treatment may be limited by personalized medicine tests used to stratify patients. Although they questioned the presumed technological imperative presented by personalized medicine technologies, they called for increased efforts to prepare the health-care system to effectively integrate these technologies. This study represents an early but important effort to explore public values toward personalized medicine. This study also provides evidence of the public's ability to form coherent judgments about a new policy issue. Concerned that personalized tests might be used to ration care, they suggested that treatment should be made available if patients wanted it, irrespective of tests that indicate little benefit. This issue raises clinical and policy challenges that may undermine the value of personalized medicine. Further efforts to deliberate with the public are warranted to inform effective, efficient and equitable translation of personalized medicine.

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Section: Data Collection and Analysismentioning

confidence: 99%

Citizens' perspectives on personalized medicine: a qualitative public deliberation study

et al. 2013

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“…The questionnaire was developed by our multidisciplinary team based on previous research 15 and a literature review. [31][32][33][34][35][36] It was pretested through 3 rounds of face-to-face cognitive interviews (n = 16) and piloted with members of the Internet panel (n = 87) to assess face and content validity and comprehension.…”

Section: Study Questionnairementioning

confidence: 99%

“…[12][13][14] When engaged in deliberation about the tensions between individual and collective interests, US and Canadian publics are generally supportive of research use of stored samples. 1,15,16 Where concrete policy options have been proposed, however, majorities favor active permissionbased approaches over passive notification/opt-out processes. 1,[17][18][19][20][21] Even genetics professionals perceive risk to secondary uses of bloodspots without permission, including reduced uptake and loss of public trust.…”

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confidence: 99%

Using Newborn Screening Bloodspots for Research: Public Preferences for Policy Options

et al. 2016

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OBJECTIVES: Retaining residual newborn screening (NBS) bloodspots for medical research remains contentious. To inform this debate, we sought to understand public preferences for, and reasons for preferring, alternative policy options. METHODS:We assessed preferences among 4 policy options for research use of residual bloodspots through a bilingual national Internet survey of a representative sample of Canadians. Fifty percent of respondents were randomly assigned to select reasons supporting these preferences. Understanding of and attitudes toward screening and research concepts, and demographics were assessed. RESULTS:Of 1102 respondents (94% participation rate; 47% completion rate), the overall preference among policy options was ask permission (67%); this option was also the most acceptable choice (80%). Assume permission was acceptable to 46%, no permission required was acceptable to 29%, and no research allowed was acceptable to 26%. The acceptability of the ask permission option was reduced among participants assigned to the reasoning exercise (84% vs 76%; P = .004). Compared with assume/no permission required, ordered logistic regression showed a significant reduction in preference for the ask permission option with greater understanding of concepts (odds ratio, 0.87; P < .001), greater confidence in science (odds ratio, 0.16; P < .001), and a perceived responsibility to contribute to research (odds ratio, 0.39; P < .001).CONCLUSIONS: Surveyed Canadians prefer that explicit permission is sought for storage and research use of NBS bloodspots. This preference was diminished when reasons supporting and opposing routine storage, and other policy options, were presented. Findings warrant consideration as NBS communities strategize to respond to shifting legislative contexts. Medical Biology, CHU de Québec, and i Molecular Biology, Medical Biochemistry and Pathology, Université Laval, Montreal, Quebec, Canada; and j Department of Family and Community Medicine, Sinai Health System, University of Toronto, Toronto, Ontario, Canada Dr Miller conceived of and led the study, designed the data collection instruments, and coordinated and supervised data collection and analysis; Dr Hayeems assisted with study design and the design of the data collection instruments, participated in the oversight of data collection and analysis, and drafted the manuscript; Drs Barg, Bombard, and Cressman assisted with study design and the design of the data collection instruments and participated in the oversight of data collection and analysis; Dr Painter-Main conducted the statistical analysis; Drs Wilson,

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“…The questionnaire included background information on the principles of screening and the varied effects of NBS (for example, early diagnosis and treatment, reproductive-risk information, false-positive results and overdiagnosis) followed by quizzes, a discrete choice experiment and a reasoning exercise. The questionnaire was developed by a multidisciplinary team on the basis of our preceding qualitative studies 19,25 and a review of the literature, [26][27][28][29][30][31] and was pretested through face-to-face cognitive interviews (over three rounds with n ¼ 16) and piloted with members of the internet panel (n ¼ 87) to assess comprehension, face and content validity.…”

Section: Questionnaire Designmentioning

confidence: 99%

Public views on participating in newborn screening using genome sequencing

et al. 2014

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Growing discussion on the use of whole-genome or exome sequencing (WG/ES) in newborn screening (NBS) has raised concerns regarding the generation of incidental information on millions of infants annually. It is unknown whether integrating WG/ES would alter public expectations regarding participation in universal NBS. We assessed public willingness to participate in NBS using WG/ES compared with current NBS. Our secondary objective was to assess the public's beliefs regarding a parental responsibility to participate in WG/ES-based NBS compared with current NBS. We examined self-reported attitudes regarding willingness to participate in NBS using a cross-sectional national survey of Canadian residents recruited through an internet panel, reflective of the Canadian population by age, gender and region. Our results showed that fewer respondents would be willing to participate in NBS using WG/ES compared with NBS using current technologies (80 vs 94%, Po0.001), or perceived a parental responsibility to participate in WG/ES-based NBS vs current NBS (30 vs 48%, Po0.001). Our findings suggest that integrating WG/ES into NBS might reduce participation, and challenge the moral authority that NBS programmes rely upon to ensure population benefits. These findings point to the need for caution in the untargeted use of WG/ES in public health contexts.

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Citizens’ Values Regarding Research With Stored Samples From Newborn Screening in Canada

Cited by 42 publications

References 37 publications

Citizens' perspectives on personalized medicine: a qualitative public deliberation study

Citizens' perspectives on personalized medicine: a qualitative public deliberation study

Using Newborn Screening Bloodspots for Research: Public Preferences for Policy Options

Public views on participating in newborn screening using genome sequencing

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