Background: Cousin marriages, in the Netherlands most frequently between Turkish or Moroccan couples, are at higher risk of having offspring with recessive disorders. Often, these couples not perceive or accept this risk, and it is hardly considered a reason to refrain from family marriages. Preconception carrier screening (PCS) is offered to Jewish groups, and more recently in the Netherlands, to genetically isolated communities. In this study, Dutch Moroccan and Turkish women’s perspectives on preconception carrier screening (PCS) and reproductive choices were explored. Methods: Individual interviews were held with Dutch Turkish and Moroccan consanguineously married women (n=10) andseven groupdiscussions with Turkishand Moroccan women(n = 86). Transcripts and notes were analyzed thematically. Results: All women welcomed PCS particularly for premarital genetic screening; regardless of possible reproductive choices, they prefer information about their future child’s health. Their perspectives on reproductive choices on the basis of screening results are diverse: refraining from having children is not an option, in vitro fertilization (IVF) combined with pre-implantation genetic diagnosis (PGD) was welcomed, while prenatal genetic diagnosis (PND), termination of pregnancy (TOP), in vitro fertilization with a donor egg cell, artificial insemination with donor sperm (AID), and adoption, were generally found to be unacceptable. Besides, not taking any special measures and preparing for the possibility of having a disabled child are also becoming optional now rather than being the default option. Conclusions: The women’s preference for PCS for premarital screening as well as their outspokenness about not marrying or even divorcing when both partners appear to be carriers is striking. Raising awareness (of consanguinity, PCS and the choice for reproductive options), and providing information, screening and counseling sensitive to this target group and their preferences are essential in the provision of effective health care. Keywords: Consanguinity, Preconception carrier screening, Reproductive choices, Premarital screening * Correspondence: firstname.lastname@example.org Department of Medical Humanities, Amsterdam Public Health research institute, School of Medical Sciences, Boelelaan 1089a, 1081, HV, Amsterdam, The Netherlands Full list of author information is available at the end of the article © The Author(s). 2018 Open Access This article is distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated. Verdonk et al. BMC Women's Health (2018) 18:79 Page 2 of 10 Background calculated, the medical and the social, the choice to In the Netherlands, preconception screening (PCS) is of- eliminate social risk gets priority. fered and studies are conducted to assess preferences for In the Netherlands, 80% of Turkish and Moroccan mi- targeted carrier screening among Jewish groups and, grants (who make up about 11% of the Dutch popula- more recently, among genetically isolated communities tion) marry a within-ethnic group partner [12, 13]. The [1, 2]. When partners carry a same genetic mutation for prevalence of consanguineous marriages among these autosomal recessive disorders they have a 1 in 4 chance groups is about 20–25% . In the general population, of having an affected child. In clinical genetics, a consan- a couple’s statistic risk of having offspring with severe guineous marriage is defined as an intra-familial union autosomal recessive (AR) genetic disorders is 2–3%, between people who are second cousins (fifth-degree rel- whereas on average consanguineous couples have an atives) or closer related family members [3, 4]. These additional 2–3% risk, thus 4–6%. However, only a mi- couples more often have identical DNA inherited from a nority (10–12%) of consanguineous couples have an in- common ancestor. Hence, they are a target group for creased risk of 25% or higher and thus the other 80% preconception carrier screening (PCS) also for rare dis- has a similar risk as non-consanguineous couples [3, 4]. eases beyond already identified and highly prevalent re- This is related to the possibility that both parents are cessive disorders such as thalassemia [3, 4]. In the carriers which is higher for consanguineous couples. The Netherlands, where this most frequently concerns cou- most common autosomal recessive (AR) hereditary dis- ples of Moroccan and Turkish descent, consanguinity is orders among Dutch ethnic minorities are haemoglobi- a sufficient indication for genetic counseling . Dissem- nopathies (HbP), such as thalassemia and sickle cell inating genome-based information to an ethnically di- disease . verse audience demands reflection on intercultural In many Middle Eastern and Mediterranean countries, communication and ethical questions. Besides, genetic PCS is offered premaritally on a large scale, especially technology can affect women differently than men as where both the prevalence of these AR disorders and women feel, and are held, more responsible for the consanguinity are relatively high [9, 16]. Although new, well-being and health of children than their partners . mostly rare, AR diseases are identified continuously, Not much is known about the perspectives of consan- screening programmes focus on a number of diseases guineously married Turkish or Moroccan women on which are highly prevalent among the population. They screening, counseling and the reproductive options and offer people the possibility of considering the risks of choices available to them. Currently, improved risk as- consanguinity for offspring and, to a lesser degree, mak- sessment tools are being developed to identify larger ing informed choices, such as taking preventive mea- numbers of pathogenic mutations (including rare muta- sures . Some forms of screening are mandatory, for tions) and differentiate between high-risk and low-risk instance the test for HIV, hepatitis and thalassaemia be- couples . For this study, we have considered the most fore civil marriage in Turkey. However, rather than pro- common reproductive options that follow upon an un- viding the results of these tests, providing proof of being favorable outcome of PCS: (1) refraining from having tested is obliged. Besides, religious marriages without children; (2) termination of pregnancy (TOP) after pre- having a civil marriage are still practiced frequently. In natal genetic diagnosis (PND); (3) in vitro fertilization Morocco genetic tests are hardly available. Nevertheless, (IVF) combined with Pre-Implantation Genetic Diagno- in an anthropological field study in Morocco and Turkey sis (PGD); (4) in vitro fertilization with a donor egg cell; , all people were very well aware of the discussion on (5) artificial insemination with donor sperm (AID); (6) family marriages and the genetic risk for offspring but adoption, or (7) not taking any special measures and/or most did not perceive and did not accept the medical preparing for the possibility of having a disabled child. risk as priority. Turkey and Morocco are both populated by Islamic Earlier research shows that in the Netherlands, Dutch majorities . An important perceived advantage of con- Turks and Moroccans find PCS acceptable for severe sanguineous marriages is the elimination of social risk. It disorders because it is perceived to provide information offers security for women and children by strengthening about the health of future children [10, 11]. Van Elderen of family ties [7–9]. Although the discussion on medical et al. explored the acceptability of PCS in general for risk for offspring in the case of marrying within the fam- Dutch Moroccans, Turks and Surinamese and they did ily is widespread, genetic literacy is low [8, 10, 11]. Be- not find an association with education, religion, or con- sides, these groups hardly perceive or accept a medical sanguinity . Thus, in general, primary prevention of risk, or do not consider that risk a reason to refrain from diseases in the form of PCS seems welcomed. Less is family marriages. Medical risk is not prioritized, as it is known, however, about how this prevention is perceived often considered just one of many risks in life or it is by Dutch Moroccan and - Turkish women and more framed in terms of fate . When both risks are specifically, consanguineously married women, and how Verdonk et al. BMC Women's Health (2018) 18:79 Page 3 of 10 they welcome reproductive choices. To our knowledge, considered responsible for procreation and care for chil- there are no studies that focused on consanguineously dren. Besides, women compared with men in general married women’s preferences with regard to PCS and have more contact with their doctors because of repro- several reproductive choices, even though more tech- ductive care. Hence, we drew from both groups and nologies are going to be available to estimate risk. Little interviewed women only with the exception of one inter- is known about how to deliver health care to particular view with a young couple. Ten in-depth interviews were migrant groups and how to accommodate to their needs, conducted by OS in Dutch to explore consanguineously but good preconception care requires insight into target married women’s perspective(s) towards (a) the possibil- groups’ perspectives, preferences and choices. Beyond ity of screening, (b) the reproductive options available diseases with a high prevalence, PCS technologies are afterwards, and (c) the contexts and embeddedness of also expected to identify carrier couples for rare AR dis- their choices. We used purposive sampling to maximize eases. With this study we aimed to gain a better under- the richness, depth and variability of the data and se- standing of the perspectives of Dutch Moroccan and lected a variety of individual interviewees, based on a Turkish women and, more specifically, consanguineously marriage with a cousin, having children or not, disabled married women among them, on what PCS is expected children or healthy, age (ranging from 24 to 50 years to offer even when they do not perceive or accept a old), and Turkish and Moroccan background (Table 1, medical discourse on risk. Therefore, exploring the pos- pseudonyms used) . Since the interviews were held sible future implications of PCS in terms of reproductive in Dutch, all women had to be able to speak Dutch suffi- and related choices is relevant, especially for these ciently. For this article, quotes from the interviews were women. translated in English. Women were recruited through Therefore, a key concept in our study is frameworks of Turkish and Moroccan women’s organizations, a choice. It indicates how people perceive choice, how self-support group for mothers with a disabled child via choices are embedded in broader contexts, and how they a care organization, and through students. During one are influenced by individuals, families and communities interview the husband was present (Meryem and and are related to socio-economic circumstances, oppor- Kerem). In such case, concern is warranted whether and tunities, belief systems and social networks . Framing how the presence of their partner influences the partici- refers to the selection of aspects of reality which are im- pants’ freedom of expression. However in this interview, portant in the interpretation, communication, and recep- both partners were very open to discussing moral di- tion of (social) knowledge. A process of framing takes lemmas and the reproductive choices they made, as place to bridge the ‘know-do’ gap, the gap between what shown in our results section. Interviews were held in the is known and what is done . Frameworks of choice is women’s homes and took approximately 2 h. not opposed to the concept of individual choice but it We were interested in the possible choices and the “puts into perspective approaches that regard ‘individual’ contexts in which meaning making on PCS and repro- or ‘autonomous choice’ [main concepts in classical bio- ductive choices is produced. Starting point is the notion ethics] as the basis of predictive and reproductive that health care decisions are made and embedded in decision-making” . Understanding the frameworks of daily life. Discussions in ‘natural’ groups of people, choice of individuals makes it possible to contextualize people who know each other already, is useful to gain their choices. Such an approach takes into account the insight in the content of social knowledge about topics women’s individual agency, their social interdependency such as consanguineous marriage, PCS, and reproductive or ‘relational autonomy’ , and the structural gen- choices, and to access how that knowledge is generated, dered cultural and religious contexts in which the framed, and embedded within possible choices. These women’s choices are embedded. In this study we explore discussions help to gain insight in an agreed upon per- Dutch Moroccan and Turkish women’s perspectives on spective on consanguineous marriage, PCS, and repro- partner choice, marriage, having children, PCS and re- ductive options shared group culture . So prior, and productive choices as embedded. running parallel (April 2011–January 2012) to the indi- vidual interviews (December 2011–September 2012), 7 Methods natural group discussions with in total 86 participants, Despite differences such as societal and ethnic back- were organized by women’s organizations (2 Turkish, 3 ground and differing interpretations of Islam, Dutch Moroccan, and 2 mixed groups) and led by EB, an an- Turkish and Moroccan women have similar characteris- thropologist with longstanding experience with women tics such as a simultaneous arrival in the Netherlands, with these background both as migrants in the class background, and patterns of partner choice . Netherlands as well as in countries of origin, and who Both groups have a relatively high rate of consanguin- had collaborated with the women’s organizations before, eous marriage and in both groups, women are and by OS (an anthropologist and a PhD-student). The Verdonk et al. BMC Women's Health (2018) 18:79 Page 4 of 10 Table 1 Overview of Individual Interviewees’ Characteristics a b No. Name Education Ethnic back-ground Migrant status Children Disability/disease (1st or 2nd generation, age of migration) 1 Nawel Highly educated Moroccan 1st generation, age 29 yes Two children diseased Genetic risk unknown 2 Kaoutar Intermediate Moroccan 1st generation, age 21 yes Child diseased Genetic risk present 3 Nesrin Highly educated Turkish 2nd generation yes Child diseased Genetic risk unknown 4 Meryem (& Kerem) Intermediate Turkish 2nd generation yes Two children diseased Genetic risk present 5 Aysel Intermediate Turkish 2nd generation no Child desired 6 Gülşen Intermediate Turkish 2nd generation yes 7 Farida Low Moroccan 1st generation yes One child diseased Genetic risk present 8 Malika Low Moroccan 1st generation yes 9 Amade Intermediate Turkish 1st generation yes 10 Sarah Intermediate Moroccan 2nd generation yes Pseudonyms Disability/disease not specified; the information is based on mothers’ evaluation of the disease and its genetic risk participants were informed about the research, but there Validity was reached by triangulation of research was no personal relationship between researchers and methods, discussions among the researchers (researcher participants. The women’s organizations had regular triangulation), in-depth interviews, and minutes of the group meetings with women from the local communi- group discussions (data triangulation). Fieldwork was ties. The women knew each other well and as they met carried out by the two anthropologists in the research regularly, they formed ‘natural groups’. Social workers team. All researchers are female. and group leaders of these organizations, community The individual interviews were tape-recorded with the members themselves knowing the population very well, participants’ consent including the use of direct quotes approached participants personally for the individual in- in the manuscript. During the group discussions, notes terviews or approached women during these regular were taken by the two researchers who were present. meetings. When the women gave permission for the They asked for permission to record the group discus- interview, the researcher contacted them. Group meet- sions, but not all women consented. New topics raised ings with the particular aim to discuss consanguinity during group discussions were checked in interviews and reproductive choice were also took place in the and vice versa and no more interviews were conducted community centers that were run by the women’s orga- when no new information emerged (data saturation) (see nizations. Not all participants of these group discussions Table 2). were consanguineously married and 2 participants had PV hand-coded the interviews line by line. Researcher other ethnic backgrounds. As the women’s organizations triangulation was applied to base coding and analytic de- are experienced with volunteering translators during cisions on convergent validation: discussions on coding, their meetings, we trusted in these translators during the clustering, and analyzing were held among all four re- group meetings for this project. Hence, a female volun- searchers. All the interviews were transcribed verbatim teer who could translate between Dutch-Arab, and read several times to get a feeling for the depth of Dutch-Tamazight, as well as a female volunteer who the data, and to collate and discuss ideas that came up could translate between Dutch-Turkish were present in during its reading. Based on a thematic analysis ap- the group discussions. The women do not have official proach, we identified key themes relating to PCS and re- interpreter credentials. In the group discussions and in- productive options and clustered thematically to identify terviews the same topic list was used which was based and report patterns and categories in the data . For on the literature. Participant observation was partly pos- instance, we identified 19 characteristics of a good hus- sible for instance during dinners with the respondents band (code), such as trustworthiness, education, and and at meetings in mosques. This offered the opportun- parents’ approval. Focusing on the research question, ity to learn about the background of the women and frameworks of choice helped to contextualize preferences seek their views in the discussions between them. for and ambivalences towards PCS and reproductive Verdonk et al. BMC Women's Health (2018) 18:79 Page 5 of 10 Table 2 List of Topics Personal information Age, educational background, migrant status, age of migration, children. Consanguinity Relationship to partner prior to marriage, marriage history, perspectives on consanguinity (disadvantages, advantages), consanguineous marriages in family and social environment, differences in perspectives among generations. Genetic risk Knowledge, perspectives on genetic risk. Children’s health Disease, diagnostic history, genetic background, living with a child with a disability/disease, mother’s and father’s ways of coping, experiences in health care. Genetic testing Perspectives on testing. Reproductive options The possibilities of PCS in the future: (1) not having children; (2) termination of pregnancy (TOP) after prenatal genetic diagnosis (PND); (3) in vitro fertilization (IVF) combined with Pre-Implantation Genetic Diagnosis (PGD); (4) in vitro fertilization with a donor egg cell; (5) artificial insemination with donor sperm (AID); (6) adoption, or; (7) not taking any special measures or preparing for possibly of having a disabled child. options. Quotes presented are mainly derived from the The ideal for all of them seems that a couple should individual interviews as they give an insight not only in be ‘able to live a life together’ or compatible, and hus- what was said, but also in ‘who the women were’ and bands should meet criteria such as an education, work, how they expressed themselves. parents’ consent, or not using drugs. Most important and always mentioned was being a Muslim as a precon- Results dition for a successful marriage: “If all men follow the We identified four major themes in the women’s per- Prophet’s example, everything goes well” (Group discus- spectives which we will subsequently describe: (1) part- sion). In case of an arranged marriage, most women ner choice; (2) marriage and having children; (3) healthy hoped to fall in love with their partners but other children, and; (4) having your own child. women fell in love with their cousins first before enter- ing marriage. Partner choice based primarily on love Partner choice seems more important for the second generation mi- Perspectives on PCS and the options which ensue are grant women than for the first generation. For all framed and embedded within the contexts of family, women, in arranged or love marriages, cousins are only religion, and gendered norms. These aspects were seen as eligible if they are men with whom the women had little mutually constituting and influencing each other. For a part contact before marriage, often because they live far away of the women consent with the rules set within these in the country of origin. ‘Real cousins’, i.e. kin that you contexts is expressed in how consanguineous marriage is actually grew up with and who ‘feel like a brother’, are ‘normal’ or even a preferable choice and how they ‘just’ not eligible to marry , and the Moroccan group leader stopped working after marriage. Other (especially younger) of one of the group discussions expresses this widely women express ambivalence towards consanguineous held belief as follows: “How to marry someone with marriages and gender roles. Aysel (26 years), grew up in the whom you played in the sandpit?” Netherlands. Originally she opposed to cousin marriage, Forced marriage was rejected by all women. But in but then developed feelings for her cousin although she practice some women had either experienced or wit- was not in love. But he just seemed ‘perfect,a ’ nd sheac- nessed both covert and overt coercion. In any case, fam- cepted his marriage proposal. Soon she considered breaking ily and parents are heavily involved in partner choice, up the relationship, continued on however, and a difficult either by arranging or consenting to a marriage. time awaited them. After a holiday and spending time to- Most women felt they could have refused a proposed gether they started getting along again. Her Turkey-raised spouse and, therefore, believe they have individual au- husband got an education in the Netherlands, started his tonomy in partner choice. The women perceive that own business, and let go of his rules for her: men are granted more freedom than women, but the younger generation seems to claim and gain more and “And since, about four years, everything is perfectly more ground for individual partner choice. Nevertheless, happy. Because otherwise I would get a divorce. approval of parents remains highly important for most When it doesn’t work, it was just a black life that I see women . before me. Just, one and a half year, a black life. I could not live like that my entire life. And he says the Marriage and having children same thing. He gave me time. He says Aysel, look, Malika finds PCS unnecessary ‘because everything is when you say ‘this does not work’ then it does not God’s will’, but all the other women were positive about work.” (Aysel). PCS, mainly to have certainty about the child’s health. Verdonk et al. BMC Women's Health (2018) 18:79 Page 6 of 10 Kaoutar, (divorced) who has a child with a severe disabil- should stop seeing each other, which is what Gülşen ity, favours PCS for a possible next child with a possible would do because: next husband: ‘When a child is ill during the night, as a mother you ‘To have a clear picture. With him [ex-husband, are completely stressed out. Measuring their cousin], I do not know what caused it. Yes, I’d just temperature, when they have diarrhoea you have to want to know.’ (Kaoutar). take care of them. And when a child will have a disease it is really not worth it to marry someone, I Furthermore, although the women desire healthy chil- would not do it. (…) They think let’s do it because it dren and intend to participate in PCS, they consider the is a small risk. But later when they realize it when genetic risk related to consanguinity to be low. Consan- they have it [the child] they regret it, because it is not guineous couples can have healthy children and worth it. It will destroy your marriage.’ non-consanguineous couples can have disabled children, they argue; other factors cause health problems too. Gen- However for Gülşen, a genetic risk up to 25% is not etic risk does not seem to play a role in consanguineous high enough to worry about, because it does not provide partner choice, and may even be denied. This does not con- enough certainty about the child’s health. Rather than flict with their interest in PCS. PCS offers the possibility to having PCS after marriage, the women do prefer pre- get information on the health of the child, about compati- marital screening and carrier status to be added to the bility with the partner, or about reproductive potential (as long list of criteria for eligible husbands. also found by e.g. ). Children are considered to be central to a marriage; refraining from having them is out of the Healthy children question because ‘you want to feel like you are a family’ If a carrier couple wants to have (more) children together, (Gülşen). Aysel literally does not want to think about the termination after PND or IVF with PGD are possible op- question: ‘All those strange questions that I cannot answer! I tions. Both technologies put a heavy burden on women, really do want a child.’ Farida, who chairs a women’s but with PND/termination, the women perceive loss, such organization and hears of illegal polygamy cases in the as transgressing religious rules, the difficulty of letting go Netherlands, explains how actively refraining from having of a pregnancy, and uncertainty about the future. With children for carrier couples can be risky for women: IVF/PGD, they perceive only gain, i.e. certainty about the child’s health. For some women, termination is taboo and ‘You have to know for sure that this man can be forbidden (‘haram’), for others, it is acceptable if the trusted, that he remains faithful. Generally, when you mother’s life is endangered or the child’s diagnosis is se- are in love, everything is okay, but after that period, vere. Religious permission is decisive but the women seem there might be a second wife, as he has a right to take uncertain about what is allowed: a second wife if he wants children.’ (Farida). ‘Abortion is the last option I would consider. I try to Therefore, not marrying a carrier partner is a viable op- live by my faith. […] I heard that before the child is tion and for some divorce is preferable to refraining from one and a half month you can have an abortion when having children. Aysel, hoping to get pregnant for several something is wrong. After that period, not anymore. years now, would not have married her cousin if they were But I have only heard this, I do not know for sure a carrier couple. For women in love marriages, breaking up now. First, I would check whether that is allowed by the relationship is not an option. Gülşen (25 years, arranged my faith. If it is allowed, I would do it.’ (Aysel). marriage) balances avoiding and accepting the risk of hav- ing a child with a disease while embracing PCS itself: Meryem and Kerem are a carrier couple, and after their first baby died from an AR disease they opted for ‘I do not think that someone, when she hears that she PND and possibly, termination, in the second pregnancy. has 25% chance to have a baby with a genetic disease, Tragically, Meryem’s second baby also died but from yet will say no, when she wants to marry. When they another AR disorder which had not been tested during really love each other. But when that woman says no, pregnancy. They went to Turkey for IVF after PGD. Ra- the risk is too large so I will not marry, that is ther than a burden, it felt a bit like a holiday to her, possible too. It can work in both ways. It is a choice in-between the treatments. Now, she has healthy twins and about risk assessment.’ (Gülşen). and states that IVF/PGD provides ‘certainty’: Being in love with your partner is considered a disad- ‘Suppose you get pregnant, and you want to terminate vantage for a carrier couple, but an arranged couple it, that is really hard. So you’d rather have a selection Verdonk et al. BMC Women's Health (2018) 18:79 Page 7 of 10 at the start, and then have it placed back. Then you gametes, complications may arise as the adopted child know that it all goes well. It is just 98, 99% certain grows up: that everything goes well.’ (Meryem and Kerem). ‘I would really love to. And so does my husband. But, In contrast to abortion, the women are certain that when I adopt a girl, after a while she is no longer […] Islam allows IVF/PGD, although Islam offers no precise halal for my husband. And then we live in the same definition of the beginning of life, the moment of con- house and she must cover herself all the time and she ception, and the onset of ensoulment. When couples do must pay attention to everything and she is just not not wish to separate, IVF/PGD as a preventive measure ours.’ (Aysel). seems acceptable because an actual pregnancy is not yet established as Gülşen explains: Both the use of a donor gamete and adoption do not result in having ‘your own child’, but whereas ambiva- ‘No, then it is not yet a child, only when the brain is lence exists towards adoption, the use of donor gametes developed and so on. Actually, from the moment of is clearly refuted. conception it is a child. But the organs are not developed yet.’ Discussion A complex mix of religious, secular, cultural and gender When a woman is pregnant, the child is accepted. logics frames the women’s perspectives on PCS and their Having an affected child is an assignment from God, not a reproductive choices. In line with earlier reports, the punishment, but rather the contrary, according to Dutch Turkish and Moroccan women in our study wel- Kaoutar: an affected child is for selected people exclu- come PCS [10, 11]. Above all, they prefer information sively, for those with sabr, patience, energy, and power. In about their future child’s health. A Dutch anthropo- the end, most women agree: ‘How we are made is God’s logical study concluded that, according to Dutch Muslim will’. theologians, imams and physicians, couples would not be interested in PCS because they either consider the genetic risk to be low or have religious reasons and Having your ‘own’ child: Biological and social parenthood choices to refrain from screening . But in close For most women, a child should be genetically one’s agreement with Dutch Muslim theological expert opin- own and born within marriage. A donor gamete is ‘not ions that in Islam, pursuing health and gaining know- your own child’. Despite the flexibility she perceives in ledge through science are important, our study reveals her faith, Farida stressed that: that the women do want to know. Second, the women’s outspokenness about not marry- ‘I do not think that anyone will do that. It is not your ing or even divorcing when both partners are carriers is real child. And it is not allowed by our faith. Whether striking, as is their preference for PCS for premarital it is a man’s or a woman’s gamete, it does not matter. screening. Marriage should not endanger the health of You must be married to have children.’ (Farida). future children . Third, the difference in attitude towards PND/termin- Nesrin, however, assumes that couples would differen- ation of pregnancy and IVF/PGD is remarkable. For tiate between egg cells and sperm. Her remark exempli- some women PND/termination is taboo and forbidden fies how the women perceive double standards for men (‘haram’). For others, it is a serious option to consider and women, not as large inequalities, but small and under rare conditions. In any case, religious permission sensible, ‘just the way it is’: is decisive. Women’s insecurity about Islamic perspec- tives towards termination of pregnancy has been re- ‘I think that for mothers, they will be less difficult, ported earlier [25, 26]. Other studies also show that because the fathers’ sperm is more important. Because termination of pregnancy is hardly acceptable to migrant he is the namegiver. So yeah, it doesn’t matter which Muslim women [10, 11, 25], although reproductive mother. If only the name of the father is continued. [I: choices including termination of pregnancy, may be And what do you think yourself?] No, I do not think highly dependent upon the particular diagnoses . so. [I: For the same reasons?] No, I would not…no. I The women were very positive about IVF/PGD, because would not like it. Even when I know it is for medical they expect it to provide certainty about a future child’s reasons, it almost sounds like adultery.’ (Nesrin). health. Interestingly, they did not mention the burden for the mother, the artificiality of conception, or the Like donor gametes, adopted children are ‘not your health risks. Rather, they stressed the fact that these pro- own’. Although adoption is preferred over donor cedures take place outside of the womb, and are Verdonk et al. BMC Women's Health (2018) 18:79 Page 8 of 10 therefore acceptable interventions and in line with sup- from other technologies. In particular contraceptive posed religious prescriptions. measures have been revolutionary in changing women’s Fourth, reproductive technologies such as PCS, IVF and men’s lives before. and PGD, are considered to be valuable particularly be- There are several limitations to our study which re- cause they are regarded by the interviewees as interven- quires further research. First, the findings must be fur- tions that provide certainty about the health of a future ther verified in larger studies. We spoke with only ten child. For our interviewees, refraining from or avoiding individual women and seven groups; partners, families, future procreation is not an option. Especially since hav- and unmarried individuals were not included. Second, as ing children is socially required; it is grounded in family found in other studies, in reality the women may be values and gender norms . more positive towards termination of pregnancy  Finally, gamete donation and adoption, are considered and more negative towards IVF/PGD . Third, effect- less acceptable because the women want to have ‘their ively conveying information about genetic technologies own child’. Not doing anything or preparing for a dis- during the interview may have been problematic because eased child are hardly discussed, since they seem to be genetic literacy is low [18, 19]. Besides, the costs of tech- the default option: when a woman is pregnant, a child is nologies in the interviews has not been discussed. accepted as it is. Fourth, all the women spoke Dutch and were prepared Our findings raise several concerns. Most of our inter- to speak with us about this subject, although we in- viewees had no individual experiences with PCS and its volved a translator in our group discussions. consequences. They emphasized their own agency but Our study has several implications. In general, more also explained how their decisions are and would be em- awareness seems useful about consanguinity, genetic bedded and framed within gender roles, religious and risk, and counseling. Misunderstanding occurs because cultural factors. Our interviewees, for instance, did men- on the one hand consanguineous couples underestimate tion faith, gender inequalities such as polygyny or the their genetic risks, although they do want to know what greater freedom that men have in choosing a partner. these risks are, while on the other hand, primary health The concept of frameworks of choice is relevant here. care providers in the Netherlands think that consanguin- On the other hand, from a health care perspective, re- eous couples do not wish to discuss these issues. In specting the women’s autonomy, means that one is addition, many primary health care providers disapprove obliged to promote it . Women are confronted with of consanguineous marriage . different reproductive choices than men and face differ- Second, information can be provided about PCS and ent consequences . Our interviewees did not mention IVF/PGD, and about the limitations that these technolo- the possibility of being refused as marriage partners; they gies have. The genetic variants that are found may have only considered the possibility of refusing; conceptions unknown effects and not all possible disorders are tested that genetic or biological ancestry is ‘stronger’ through for, but people may feel reassured of having a healthy paternal than maternal lines, may explain their views baby after PCS and/or IVF/PGD. [29, 30]. But carrier status may stigmatize women in par- Third, PND/termination was mostly refuted because of ticular, labelling them as non-eligible wedding partners religious beliefs, but health care providers should still dis- within their communities . When screening and the cuss it [25, 32]. Different Islamic viewpoints do exist about disclosure of the results cause problems for women with termination of pregnancy, which may be mentioned by positive carrier status, they face either the risk of having family physicians or theological experts . For instance, a child with a disability, or of being an unmarried out- according to some Islamic scholars termination is permis- sider. The women we interviewed do not expect prob- sible in case the child has a severe condition. lems, possibly because healthy children are so important Finally, our study has shown that PCS seems wel- to the family. But not marrying or divorcing means that comed in particular before marriage. Health services the women have to share their genetic information with can help individuals to make choices in the view of their families. Pakistani adults in the UK would not their specific risks and frames of choices as related to readily share genetic information within the family as family goals, ethical and religious values, and to act Shaw and Hurst  explain; information was kept pri- in a manner which supports and confirms their vate for reasons related to disruptive effects. In our choices [4, 29, 33]. study, the women did not refer to these consequences of However, implementing premarital screening is chal- disclosing genetic information. And besides possible lenging. Generally, couples-to-be do not present them- stigmatization, people also individually need to come to selves as such to health care providers. Both health care terms with being a carrier . New reproductive tech- providers and consanguineous couples themselves con- nologies may change the women’s social interdepend- sider the genetic risk to be low. Moreover, offering pre- ency and their social embeddedness as is already known marital screening largely relies on the subjects Verdonk et al. BMC Women's Health (2018) 18:79 Page 9 of 10 identifying their own ethnicity and consanguinity, which for this type of study in the Netherlands according to Dutch legislation. In the study we analyzed voluntary interviews with migrant women and does not necessarily correspond to genetic risk . groups of women, not an intervention with patients. Nevertheless, the study Finally, at this moment, the stigmatization of consan- has to adhere to privacy legislation which it did. No written informed guineous couples is a realistic negative side-effect of consent was obtained, as it is common that members of minority communities often decline signing consent forms. Instead of creating trust the ethnically-targeted implementation of screening based on personal relationships, forms can actually contribute to distrust, as [4, 29, 35]. Currently, Dutch politicians overestimate many people with a migrant background have adverse experiences with the risk and negatively frame consanguinity, genetic health care systems in their countries of origin as well as in the Netherlands. Furthermore, many women in particular first generation migrants are risk, migration, and relate these issues to forced mar- illiterate. The women gave their oral consent and could withdraw at any time. riages . A realistic understanding of these risks, Participants were informed about the voluntariness of the participation and the choices which can be made and the perceived that only members of the research team would have access to the interview data. Also, participants did not feel compelled to stay for the whole group benefits of consanguinity should therefore be encour- session. Nevertheless, the women gave oral consent for participating and were aged among politicians and policymakers. actively engaged in giving information. We confirm all patient/personal identifiers have been removed or disguised so the patient/person(s) described are not identifiable and cannot be identified through the details of the story Conclusion (see Medical Research Involving Human Subjects Act 1998, download from New technologies for PCS are welcomed by consanguine- http://wetten.overheid.nl/BWBR0009408/2017-03-01 and http://www.ccmo.nl/ ously married women regardless of possible reproductive en/medical-scientific-research-and-the-wmo, 18 May 2018). options because it provides information about the future Competing interests child’s health. Their preference for PCS for premarital The authors declare that they have no competing interests. screening as well as their outspokenness about not marry- ing or even divorcing when both partners appear to be Publisher’sNote carriers is striking. Raising awareness (of both risks and Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations. options and choices that can be made), and providing in- formation, screening and counseling sensitive to this tar- Author details get group and their preferences are important. Department of Medical Humanities, Amsterdam Public Health research institute, School of Medical Sciences, Boelelaan 1089a, 1081, HV, Amsterdam, Abbreviations The Netherlands. Department of Medical Humanities, Amsterdam Public AR: Autosomal Recessive; IVF: In Vitro Fertilization; PCS: Preconception Carrier Health research institute, Boelelaan 1089a, 1081, HV, Amsterdam, The Screening; PGD: Preimplantation Genetic Diagnosis; PND: Prenatal Diagnosis; Netherlands. Department of Social and Cultural Anthropology, VU University, TOP: Termination of Pregnancy De Boelelaan 1105, 1081, HV, Amsterdam, The Netherlands. MOVISIE Netherlands Centre for Social Development, Catharijnesingel 47, 3511, GC, Acknowledgements Utrecht, The Netherlands. The researchers thank the anonymous reviewers and dr. Marieke Teeuw and prof.dr. Leo ten Kate for their expertise, the women’s organizations and, of Received: 21 August 2017 Accepted: 21 May 2018 course, the women (and one husband) who shared their stories so openly and frankly. References Funding 1. Holtkamp KCA, Van Maarle MC, Schouten MJE, Dondorp WJ, Lakeman P, The study was funded by CSG Centre for Society and the Life Sciences. The Henneman L. Do people from the Jewish community prefer ancestry-based funding agency had neither a role in designing, collecting, analyzing and or pan-ethnic expanded carrier screening? Eur J Hum Genet. 2016;24:171–7. interpreting the data nor in writing the manuscript. 1018–4813/16. 2. 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Published: May 31, 2018
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