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AJOB Empirical Bioethics

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Noninvasive Prenatal Testing: Views of Canadian Pregnant Women and Their Partners Regarding Pressure and Societal Concerns

Vardit Ravitsky, Stanislav Birko, Jessica Le Clerc-Blain, Hazar Haidar, Aliya O. Affdal, Marie-Ève Lemoine, Charles Dupras & Anne-Marie Laberge

To cite this article: Vardit Ravitsky, Stanislav Birko, Jessica Le Clerc-Blain, Hazar Haidar, Aliya O. Affdal, Marie-Ève Lemoine, Charles Dupras & Anne-Marie Laberge (2021) Noninvasive Prenatal Testing: Views of Canadian Pregnant Women and Their Partners Regarding Pressure and Societal Concerns, A JOB Empirical Bioethics, 12:1, 53-62, DOI: 10.1080/23294515.2020.1829173

To link to this article: https://doi.org/10.1080/23294515.2020.1829173

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Noninvasive Prenatal Testing: Views of Canadian Pregnant Women and Their Partners Regarding Pressure and Societal Concerns

Vardit Ravitskya , Stanislav Birkoa , Jessica Le Clerc-Blainb, Hazar Haidarc, Aliya O. Affdala , Marie-�Eve Lemoinea , Charles Duprasd , and Anne-Marie Labergeb

aBioethics, Department of Social and Preventative Medicine, School of Public Health, Universit�e de Montr�eal, Montreal, Quebec, Canada; bMetabolic and Cardiovascular Health, CHU Sainte-Justine Research Center, Montreal, Quebec, Canada; cInstitute for Health and Social Policy, McGill University, Montreal, Quebec, Canada; dCentre of Genomics and Policy, McGill University, Montreal, Quebec, Canada

ABSTRACT Background: Noninvasive prenatal testing (NIPT) provides important benefits yet raises eth- ical concerns. We surveyed Canadian pregnant women and their partners to explore their views regarding pressure to test and terminate a pregnancy, as well as other societal impacts that may result from the routinization of NIPT. Methods: A questionnaire was offered (March 2015 to July 2016) to pregnant women and their partners at five healthcare facilities in four Canadian provinces. Results: 882 pregnant women and 395 partners completed the survey. 64% of women anticipated feeling no pressure to take the test if it were offered routinely, and 39% were not concerned about routinization leading to increased pressure to terminate a pregnancy of a fetus with Down Syndrome. Regarding other social concerns possibly resulting from routinization, pregnant women were most concerned regarding a reduction in resources available for people with Down Syndrome and their families and least concerned regarding a decrease in the population of people with Down Syndrome. Conclusions: Our findings reflect the concerns expressed by pregnant women and their partners, both personal (pressure to test, pressure to terminate) and societal (e.g., regarding potential negative impact on people with disabilities and their families). Even if most women were not concerned about feeling pressured to test due to NIPT routinization, a large minority express concerns that should not be taken lightly. Moreover, a majority of respondents were concerned regarding pressure to terminate pregnancies due to NIPT rou- tinization as well as regarding most societal impacts they were queried on, especially the possible future reduction in resources available for people with DS and their families. Canadian policy-makers should consider these potential negative ramifications of NIPT and ensure that appropriate social policies accompany its implementation.

KEYWORDS Eugenics; NIPT; prenatal screening; pressure to test; routinization; social impact

Introduction

Noninvasive prenatal testing (NIPT) tests cell-free DNA (cfDNA) originating from the placenta and cir- culating in maternal blood in order to screen for gen- etic conditions in the fetus. NIPT provides important benefits for pregnant women and their families when compared to conventional screening and amniocen- tesis. It can be performed early in the pregnancy (week 10–11), has better detection rates for trisomies 21, 18, and 13 than current screening methods, and carries no risk of miscarriage (Griffin et al. 2018). Currently, NIPT targets mainly trisomy 21 (Down

syndrome, DS), 18, and 13. It is also widely used to screen for sex chromosome abnormalities and some microdeletion syndromes (Wapner et al. 2015). Expanded use of NIPT for a host of other conditions raises concerns related to what conditions should and should not be screened for (Norton 2016).

The test’s characteristics raise concerns regarding its routinization possibly leading to increased pressures on women to test and even terminate affected pregnancies (Kater-Kuipers et al. 2018, Dupras et al. 2018, Griffin et al. 2018, Haidar, Dupras, and Ravitsky 2016, Lewis, Silcock, and Chitty 2013, Skirton and Patch 2013, Wright and Burton 2009, Hewison 2015, Benn, Cuckle,

� 2020 Taylor & Francis Group, LLC

CONTACT Stanislav Birko [email protected] Bioethics, Department of Social and Preventative Medicine, School of Public Health, Universit�e de Montr�eal, 7101 av du Parc #3103-1, Montr�eal, QC H3N 1X9, Canada.

Supplemental data for this article is available online at https://doi.org/10.1080/23294515.2020.1829173

AJOB EMPIRICAL BIOETHICS 2021, VOL. 12, NO. 1, 53–62 https://doi.org/10.1080/23294515.2020.1829173

and Pergament 2013, Hill et al. 2013, Deans and Newson 2012, Seavilleklein 2009, How et al. 2019, Kellogg et al. 2014, Verweij, Oepkes, and de Boer 2013, Benn and Chapman 2010, Kent 2008). These pressures can range from explicit coercion on the part of health- care providers to subtle influences of prevailing social norms. Concerns are raised regarding increased rates of termination leading to eugenic social attitudes, nega- tively impacting persons living with DS and their fami- lies, increasing discrimination, decreasing support for families with affected children, increasing stigmatization, decreasing acceptance of children with disabilities, and possibly even reducing the number of persons born with DS (Dupras et al. 2018, Griffin et al. 2018, Tischler et al. 2011, van Schendel et al. 2014, Kellogg et al. 2014, Farrimond and Kelly 2011, Kaposy 2013, Lewis, Silcock, and Chitty 2013, Benn and Chapman 2010, Hall, Bostanci, and John 2009, Wilkinson 2015) .

The pan-Canadian PEGASUS study (http://pegasus- pegase.ca/) explored the implementation of NIPT into the Canadian healthcare system, including its ethical, legal, and social aspects. As a part of this project, we conducted a survey (Laberge et al. 2019, Birko et al. 2019) examining the views of Canadian pregnant women and their partners toward NIPT. A section of the survey explored Canadian pregnant women’s and their partners’ concerns regarding increased pressure to test and terminate due to the test’s routinization, as well as NIPT’s potential social impacts discussed in the bioethics literature. This paper presents and dis- cusses this subset of the survey results.

Methodology

Questionnaires for pregnant women and their part- ners were developed based on a review of the relevant literature and extraction of major themes (HH), clin- ical experience of two authors (JBL and AML), and questionnaires used in previous studies (Farrimond and Kelly 2011, Horsting et al. 2014, van den Heuvel et al. 2010, van Schendel et al. 2014, Verweij, Oepkes, de Vries, et al. 2013, Yotsumoto et al. 2012, Lewis, Hill, and Chitty 2014, Sayres, Allyse, and Cho 2012). The questionnaire for pregnant women was reviewed by the PEGASUS team for content validity and feasi- bility and was then piloted on eight women of repro- ductive age, followed by cognitive debriefing. Based on pilot results, the questionnaire was modified for clarity and length. The questionnaire for partners was adapted from the questionnaire for pregnant women.

The pregnant women’s (41 questions, Appendix 1) and partners’ (43 questions, Appendix 2) questionnaires

explored the following themes: knowledge about DS and NIPT1, written consent (results reported in Birko et al. 2019), NIPT’s clinical uses, decision-making and others’ involvement (results reported in Laberge et al. 2019), NIPT’s social impact (present paper), and future uses of NIPT. Question formats included Likert scales, “true or false” statements, multiple choice, and ranking. The questionnaire was distributed along with an infor- mation sheet explaining the differences between mater- nal serum screening, amniocentesis, and NIPT; and giving brief descriptions of procedures, test timing, risk for pregnancy, accuracy, nature of test (screening vs diagnostic), potential results, and potential outcomes.

The survey was open from March 2015 to July 2016. It was offered in English and French online and on paper (in Ontario and Qu�ebec, only in English on paper in the Western provinces). Recruitment occurred at five facilities in four Canadian provinces (British Columbia, Alberta, Ontario, and Quebec). Ethics approval was obtained from the CHU Sainte- Justine associated with the University of Montreal as well as, locally, from the CRCHU de Qu�ebec, the Ottawa Hospital Research Institute, BC Children’s Hospital, and the University of Calgary.

To explore the possible influences on views toward NIPT, participants’ responses were analyzed based on their socio-demographic characteristics, their stated intended use of test results, and knowledge of DS and available testing options. Statistical analysis was performed using Pearson Chi-Square tests, Kruskal- Wallis and Mann-Whitney U tests, Kendall’s tau cor- relations where applicable, done with IBM SPSS 24. To account for multiple testing, p< 0.001 was estab- lished a priori as the threshold of statistical signifi- cance reported. Quantitative results are presented as frequencies of responses (%).

Results

Eight-hundred-and-eighty-two pregnant women and 395 partners answered the questionnaire. Demographic characteristics of participants are described in Table 1. Assuming 380,000 annual births in Canada, this yields a maximum margin of error of 3.29% for the pregnant women and 4.93% for the partners (Statistics Canada 2019, Gregoire and Affleck 2018). One center did not keep track of women invited to participate. In four other centers (British Columbia, Ontario, and 2 in Qu�ebec), 1603 pregnant women were invited to participate. Of

1These questions were asked before the respondents were referred to the information sheet.

54 V. RAVITSKY ET AL.

these, 755 completed the questionnaire, yielding a response rate of 47.1%. All those invited were given a survey for their partner. 366 partners (from these

4 centers) completed the survey, yielding a response rate of 22.8%, assuming that all participating preg- nant women invited their partner.

Table 1. Respondents’ characteristics. Pregnant Women and Partners

Characteristic No. (%) of Pregnant Women n¼ 882 No. (%) of Partners n¼ 395

Age Mean (SD) 32.3 (4.8) 33.5 (5.7)

Gender (of partner) Other than male N/A 11 (2.8)

Province of Residence BC 251 (28.5) 24 (6.1) AB 116 (13.2) 28 (7.1) MB 2 (0.2) 0 ON 118 (13.4) 78 (19.7) QC 387 (43.9) 264 (66.8) Atlantic provinces 2 (0.2) 0 Territories (Nunavut/NWT/Yukon) 2 (0.2) 0

Country of birth Canada 708 (80.3) 342 (86.8)

Language Mostly Spoken at Home English 478 (54.2) 134 (33.9) French 355 (40.2) 257 (65.1) Other 37 (4.2) 12 (3.0)

Race/ethnicity Caucasian/white 718 (81.4) 340 (86.1) North American Aboriginal (First Nation, Inuit, Metis)

14 (1.6) 9 (2.3)

Other 150 (17.0) 46 (11.6) Religion/culture Christian 492 (55.8) 237 (60.0) Muslim 19 (2.2) 8 (2.0) Buddhist 16 (1.8) 3 (0.8) Jewish 12 (1.4) 0 Hindu 7 (0.8) 0 Sikh 5 (0.6) 2 (0.5) None/agnostic/atheist/spiritual 352 (39.9) 151 (38.2)

Importance accorded to religion/spirituality (1-5) Mean (SD) 2.26 (1.25) 1.92 (1.19)

Highest completed education level Elementary School 2 (0.2 ) 5 (1.3 ) High School 62 (7.0 ) 57 (14.6 ) Trade School 47 (5.3 ) 50 (12.8 ) CEGEP/College 175 (19.8 ) 74 (18.9 ) University 590 (66.9 ) 205 (52.4 )

Are you a Healthcare Professional: Yes 233 (26.5 ) 30 (7.8 ) Relationship Status Married 511 (57.9 ) 158 (40.0 ) Common-law 334 (37.9 ) 213 (53.9 ) Single 23 (2.6 ) 10 (2.5 ) Divorced/separated 3 (0.3 ) 2 (0.5 ) Other 4 (0.5 ) 4 (1.0 )

Already has a child 404 (45.8 ) 151 (39.0 ) With Down Syndrome 5 (0.6 ) 1 (0.3 ) With physical or intellectual disability 9 (1.0 ) 1 (0.3 )

Does anyone close to you have a child with Down Syndrome

68 (7.7 ) 35 (9.0 )

Current pregnancy is Low-risk for DS 565 (64.1 ) 235 (60.1 ) High-risk for DS 99 (11.2 ) 43 (11.0 ) Unsure 207 (23.5 ) 113 (28.9 )

Current pregnancy was conceived Naturally 790 (89.6 ) 356 (91.3 ) Using IVF 48 (5.4 ) 17 (4.4 ) Using ART other than IVF 24 (2.7 ) 17 (4.4 )

Has had prenatal screening in a previous pregnancy

284 (32.5 ) 99 (25.7 )

Has had prenatal diagnosis (chorionic villus sampling or amniocentesis) in a previous pregnancy

58 (6.7 ) 32 (8.2 )

AJOB EMPIRICAL BIOETHICS 55

Concerns regarding pressure to test or terminate

The survey explored whether the routinization and public funding of NIPT would cause women to be concerned regarding pressure to use the technology or to terminate an affected pregnancy. Respondents were asked whether they anticipated feeling personally pres- sured to test if it became routine, and whether they were concerned that women in general (societally) would feel increased pressure to test or terminate.

Pressure to test 63.9% said they would personally feel no pressure to use NIPT due to routinization and 2.8% said they would feel a lot of pressure (Figure 1). These numbers changed only slightly when asked about such pressure societally (Figure 1).

When partners were asked about routinization of NIPT causing their pregnant partner to feel pressure to test, their responses were significantly different. Partners were more concerned about NIPT causing pressure on the pregnant woman to test (Figure 1). They were, however, less concerned regarding them- selves feeling increased pressure to test (Figure 1).

Pressure to terminate 38.7% of pregnant women were not concerned regarding routinization causing increased pressure to terminate in the case of a Down syndrome diagnosis (Figure 2). More were very concerned (5 on the Likert scale) about

increased pressure to terminate an affected pregnancy than very concerned about increased pressure to test. All women who were very concerned about pressure to test had at least a moderate level of concern about pressure to terminate (at least 3 on the 5-point scale).

Views of societal concerns regarding impact on people with disabilities

The impact of routinization and public funding of NIPT on women’s views regarding several societal concerns are summarized in Figure 3. Women were least concerned about the possibility of a decreasing population of people with DS. Women were most concerned about possible reduction in resources available for people with DS and their families. A higher proportion was very concerned about negative impacts on families than about people being less willing to accept children with disabilities. No sig- nificant difference was detected between pregnant women’s and partners’ societal concerns.

Correlations

The survey included a question regarding the reason for being interested in knowing whether the fetus had DS. The response frequency is shown in Table 2. These reasons correlated with all categories of con- cern, both personal and societal (p< 0.001). Respondents who stated that they would consider

Figure 1. Levels of anticipated pressure to test due to NIPT routinization.

56 V. RAVITSKY ET AL.

Figure 3. Levels of concern expressed by pregnant women over societal outcomes of NIPT routinization.

Table 2. “Information about a baby having Down syndrome (DS) can have different uses for different people. Why are you inter- ested in knowing whether your baby has DS?”

% of women

I want to know in advance to prepare for the birth of a baby with DS if the baby is diagnosed with DS 27.1 I would consider terminating the pregnancy if the baby was diagnosed with DS 52.9 I’m unsure 14.3 I do not want to know 2.3

Figure 2. Levels of anticipated pressure to terminate a pregnancy due to NIPT routinization.

AJOB EMPIRICAL BIOETHICS 57

terminating the pregnancy in case of DS showed least concern, and those who did not want to know whether their fetus has DS showed most concern.

Correctly answering the knowledge questions regarding DS correlated only with societal concern over increased pressure to test (Pearson’s R¼-0.181), with those answering more questions correctly being less concerned. Correctly answering questions regard- ing prenatal testing correlated with societal concern regarding pressures to test (R¼-0.175) and terminate(R¼-0.152), as well as regarding a declining population of persons with DS (R¼-0.169). It bears noting that correctly answering questions regarding prenatal testing (but not DS) correlated with respond- ents’ level of education (R¼ 0.213).

Level of religiosity did not correlate with concern over personally being pressured to test, but correlated with all societal concerns (R between 0.147 and 0.227), except that of decreasing resources available for persons living with DS and their families.

In addition, concern over being personally pres- sured to test positively correlated with all societal con- cerns (R between 0.226 and 0.378).

Discussion

An interesting finding regarding women’s views of NIPT is that the majority did not expect routinization of the test to make them feel pressured to use it, nor was the majority at all concerned regarding societal pressure to test due to routinization. While this may seem to not align with the extensive bioethics literature about routinization of NIPT as raising these concerns, it can be explained by the notion that women are not concerned about feeling pressure themselves, while they are concerned about other women feeling pressure. This would also explain why partners were more con- cerned than pregnant women. Notwithstanding these findings, it is important to highlight that a strong minority (over a third of the respondents) were at least somewhat concerned about pressure to test, both per- sonally and societally, and almost half were concerned about the decrease in the population of persons with DS. Moreover, the majority of respondents were at least somewhat concerned about all the other outcomes of routinization that they were queried about: pressure to terminate, less willingness to accept disability, and a decrease in resources and negative impact on those liv- ing with DS and their families. A further major conclu- sion of this study is that Canadian women’s biggest societal concern was a possible reduction in resources for persons with DS and their families as a consequence of

the routinization of NIPT, a concern shared by experts surveyed in previous studies (Dupras et al. 2020).

Low level of concern over routinization as leading to pressure to test

The study’s finding that most Canadian women do not expect to feel any pressure at all to test if NIPT became routine contradicts the expectation expressed in the lit- erature that increased pressure to test is a key concern regarding NIPT’s implementation (Dupras et al. 2018; Lewis, Silcock, and Chitty 2013, Griffin et al. 2018, Kelly and Farrimond 2012, Birko et al. 2018, Ravitsky 2017, Haidar, Dupras, and Ravitsky 2016). It is possible that women’s low levels of hypothetical concern emerg- ing from this study may not reflect the reality of pre- natal testing, considering that systemic pressure to test and terminate is widely reported (Seavilleklein 2009, Ahmed et al. 2017, van Schendel et al. 2014).

Respondents’ low level of concern regarding pressure to test may be explained in at least two ways. First, when faced with a choice regarding testing, women may feel immune to such pressure and empowered to make autonomous decisions based on their own values. Alternatively, women may be so used to such pressure that they do not recognize or acknowledge it and thus report not being concerned, whether individually or societally. Health psychology scholars have demon- strated that “much human behavior is automatic, cued by environmental stimuli, resulting in actions that are largely unaccompanied by conscious reflection” (Marteau, Hollands, and Fletcher 2012), guided by a “nonconscious, impulsive, associative system” (Sheeran, Gollwitzer, and Bargh 2013). Others attempt to explain this kind of behavior as, e.g., reproducing “taken-for- granted social expectations” (Butler 2005), “taken-for- granted social norms” (Sumerau 2017), “internalized norms” (Foucault 2012), or “social defaults” (Huh, Vosgerau, and Morewedge 2014, Smith, Goldstein, and Johnson 2013). It may be particularly difficult to become aware of social norms when they are shaped by gendered patterns, heteronormativity, and ableism, to name a few (Butler 2005, Collins 2004, Chambers and Armitage 2009, Goodley 2018). Lippman argued long ago (1991) that social, political and economic constraints of wom- en’s lives are hidden by presenting prenatal testing alter- natives as “reproductive choices,” despite the fact they are defined by “biomedical and political systems.”

Women’s views regarding pressure felt personally did correlate with their societal concerns. One may argue that for some people such societal concerns

58 V. RAVITSKY ET AL.

may discourage testing and terminating, in order to counter (consciously or not) eugenic tendencies.

While women’s relative lack of concern is note- worthy, more than a third of women and half of part- ners anticipated routinization would make them feel pressured to test. This group cannot be ignored. Nor can we ignore the women who said increased pressure to test as an outcome of routinization would be a con- cern to them societally, or the majority of women who are concerned about increased pressure to ter- minate following a diagnosis of DS. The numbers of those concerned invite healthcare professionals, and our society, to reflect about the possible implications of implementing an easy and safe prenatal test.

The correlations between better knowledge of DS and prenatal testing, and greater concerns regarding pressure to test or terminate, are illuminating. Those not at all concerned about pressure to test (societally) had the highest level of knowledge about DS and pre- natal testing, which could indicate that more know- ledgeable people feel more immune to societal pressure. It could also indicate that better access to information can diminish concerns regarding pressure. A surprising finding of this study was that level of formal education correlated with greater knowledge regarding prenatal testing but not of DS. Future research to corroborate (or refute) this finding as well as to evaluate the pos- sible causes of this discrepancy could be worthwhile.

Future research is needed to explore the questions raised by our findings. What causes women and partners to anticipate “pressure” if NIPT becomes routine prac- tice? Do they feel that certain technologies must be used because they exist (Hofmann 2002, McCoyd 2010)? Do they feel that public funding sends a message that people should test? Do they fear pressure exerted by healthcare professionals? Do they fear being part of a decreasing minority of families raising children with disabilities? These questions call for qualitative studies exploring these concerns and their impact on decision-making.

Reduced resources for persons with Down Syndrome is the greatest societal concern

Respondents were more concerned about reduced resources for and negative impact on persons with DS and their families, than they were about a decreasing population of persons with DS. This means that some respondents who were unconcerned by a decreasing population of persons with DS, were concerned with the well-being of families raising children with DS. This could be explained in at least two ways. First, these respondents may have thought that prenatal

testing would not cause a decrease in the population of persons with DS2. Alternatively, they may have thought that the decreased number of persons born with DS will still warrant concerns regarding social policies meant to protect them and their families.

NIPT routinization causing a decrease in the num- ber of persons born with DS was least concerning, with a majority not concerned at all about this pos- sible outcome. It remains unclear whether this is because participants thought this would not happen, or because they thought it would not be worrisome. This is a limitation of our survey, which calls for fur- ther research to elucidate this point. Respondents were more concerned regarding reduced resources and negative impact than about less acceptance of children with disabilities. This could indicate that aspects determined at a public policy level (such as the resources dedicated to supporting families raising a child with DS, or protection against social discrim- ination) raise more concerns than aspects determined by individual attitudes and behaviors (such as accept- ance of children with disabilities, or decisions about terminating diagnosed pregnancies).

Knowledge regarding prenatal testing (but not regarding DS) only correlated (negatively) with con- cern regarding decreasing populations of persons with DS. This can be interpreted in at least two ways: (1) those with more knowledge of prenatal testing think that routine use of NIPT will decrease the prevalence of DS but are not concerned about this; or (2) they do not think more accurate testing will reduce the prevalence of DS. A limitation of the study is that questions gauging participants’ knowledge of Down Syndrome and prenatal testing did not include any- thing regarding the lived experience of parenting a child with Down Syndrome, but focused only on the biomedical dimensions.

Those not wishing to know whether their fetus had DS were more concerned societally. This may signal that those willing to parent a child with DS have heightened sensitivity to the possible impact of more people testing and terminating. Put simply, those con- sidering the possibility of raising a child with DS probably wish to live in a society accepting of their child. Alternatively, those wishing not to know may be concerned about societal blame on parents for bearing children with disabilities. In contrast, those considering termination expressed less societal con- cern. The fact they were less concerned regarding a decreased willingness to accept disability could be

2This aligns with a current study showing that termination rates following screening for DS by NIPT did not change (Hill et al. 2017).

AJOB EMPIRICAL BIOETHICS 59

reflecting their own choice not to raise such children. Their lower level of concern regarding negative impact on people with disabilities could indicate that those who do not wish to raise a child with DS may acknowledge to a lesser degree the societal issues faced by families raising children with disabilities.

Strengths and limitations

We report on the first pan-Canadian survey including pregnant women and partners’ concerns regarding the implementation of NIPT. Our choice of p< 0.001 as a threshold of significance reduces the risk of overesti- mating the significance of findings. Besides the study’s limitations mentioned throughout the discussion, it is possible that participants self-selected, which could affect the representativeness of the findings. Pregnant women with certain characteristics could have been more likely to recruit their partners which could account for partners showing more concern about pressure. Some similarity between pregnant women’s and partners’ responses may be due to answering the questionnaire together. The sample size and geograph- ical distribution of recruitment sites may mitigate these limitations. An important limitation of reporting such surveys is the possible confusion that the public’s views are representative of, or should determine, what is ethically acceptable. While information on public attitudes regarding health technology can be helpful to policy-makers in terms of identifying concerns, under no circumstances can particular attitudes be used to justify eroding protections for members of minority groups, in this case disabled individuals.

Conclusion

As the landscape of NIPT is shifting rapidly and as its clinical use is expanding to an increasing number of conditions, it is imperative to assess its individual and societal implications. Our findings suggest that the implementation of NIPT into the Canadian context needs to take into consideration pregnant women’s and their partners’ concerns, both personal (pressure to test, pressure to terminate) and societal concerns regarding potential negative impact on people with disabilities and their families. Even if most women are not concerned about pressure to test due to the routine offer of NIPT, the number of those who are concerned should not be taken lightly. Moreover, other concerns that do worry most of those surveyed should also be considered when designing policies and clinical norms. Canadian policy- makers should therefore consider the potential negative

ramifications of NIPT and ensure that appropriate social policies accompany its implementation. They should ensure support systems for families raising children with tested conditions and mechanisms for informed consent to protect women from pressure to test, even if this con- cern is only expressed by a minority of pregnant women. Finally, qualitative research approaches could offer deeper understanding of the causes for concern (or lack thereof) expressed by pregnant women and partners in this survey.

Acknowledgements

The authors would like to thank members of the NIPT research team: Stephanie Cloutier, Hortense Gallois, Cynthia Henriksen, Chris Kaposy, Tierry Morel-Laforce, and Marie-Christine Roy, as well as France L�egar�e, William Ehman, François Rousseau, Brenda Wilson, Marsha Michie, Talya Miron-Shatz, and Lyn Chitty.

Author contributions

The study was conceived by VR and AML, designed by VR, AML, JLB, HH. Data acquisition was overseen by SB and AA. Data was interpreted by SB, VR, AML, MEL, and CD. VR wrote the first draft in close collaboration with AML, with extensive support from SB. All authors revised the paper critically with important intellectual contributions, and approved the final version for publication. VR and AML are equal contributors.

Disclosure statement

The authors have no conflicts of interest to declare.

Ethical approval

This study was approved by the institutional review board(s) at the CHU Sainte-Justine associated with the University of Montreal as well as, locally, from the CRCHU de Qu�ebec, the Ottawa Hospital Research Institute, BC Children’s Hospital, and the University of Calgary.

Funding

VR and AML are investigators in the PEGASUS Research Project (PErsonalized Genomics for prenatal Aneuploidy Screening USing maternal blood) that was funded by Genome Canada, Genome Quebec and the Canadian Institutes for Health Research. PEGASUS received co-funding or in-kind funding from private corporations which either offered com- mercial NIPT tests (Ariosa Diagnostics Inc, San Jose, CA) or reagents and/or equipment that can be used to perform NIPT assays (Life Technologies Inc, NY, USA; Illumina, San Diego, CA, USA; QIAGEN, Hilden, GER; Perkin Elmer, Waltham, MASS, USA). This funding was at arm’s length from the

60 V. RAVITSKY ET AL.

scientific component of the project. The R�eseau de M�edecine G�en�etique Appliqu�ee (RMGA), a research network funded by the Fonds de Recherche du Qu�ebec – Sant�e (FRQS) has also provided co-funding. AML also has a Canadian Institutes of Health Research (CIHR) New Investigator Salary Support Grant, and received a FRQS Starting Grant for New Investigators – Junior 1.

ORCID

Vardit Ravitsky http://orcid.org/0000-0002-7080-8801 Stanislav Birko http://orcid.org/0000-0002-7715-6893 Aliya O. Affdal http://orcid.org/0000-0001-8924-4308 Marie-�Eve Lemoine http://orcid.org/0000-0003- 3780-0864 Charles Dupras http://orcid.org/0000-0002-9194-3252 Anne-Marie Laberge http://orcid.org/0000-0003- 1213-8288

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