To be the Parents of Children of One Sex: The Desire for a Child of the Other Sex, Mental Health, and Dyadic Adjustment of Parents who Apply for Pre-implantation Sex Selection

The desire to determine the sex of one's offspring has accompanied humankind since time immemorial. Advances in medical technology, specifically Pre-implantation Genetic Diagnosis (PGD), now enable sex selection with almost 100 percent accuracy. The use of PGD to select the sex of a fetus, in circumstances other than those dictated by medical grounds, raises complicated ethical, professional, and social dilemmas. At the center of the bioethical discussion is the issue of whether parents should be allowed to choose the sex of their children. In Israel, in contrast to most Western countries, where the use of PGD for non-medical related sex selection is banned, a unique arrangement was established in 2005. Under this arrangement, couples with four children (or, in rare circumstances, three children or less) of one sex and none of the opposite sex may apply to "The National Committee for Sex Selection through PGD" (hereinafter - the Committee), designated by the Ministry of Health to assess and approve active sex selection. Between 2005 and 2016, 784 applications were submitted to the Committee, 125 (15.9%) of which were approved. Despite the centrality of a child's gender to the parental experience, little research attention has been given to understanding the psychosocial implications of the lack of a child of the desired sex for parents and, more specifically, for mothers. Thus, applicants to the Committee constitute a "test case" for studying this subject in Israel and around the world.

The current study was based on Lazarus and Folkman's Stress and Coping Theory (1984). The conceptual model of the study (cognitive perception regarding the lack of a child of the desired sex at the time of applying to the Committee), and adjustment (mental health and dyadic adjustment) several years later (at the time of data collection). Within the context of this relationship, it also analyzed the contribution of coping styles (dyadic coping), coping resources (perceived self-efficacy, perceived social support), and the Committee's decision group. The study was conducted with the Jewish women who, with their husbands, had applied to the Committee.

As a first-of-its-kind study, this research opens a window into the world of mothers longing for a child of the missing sex, and may be a first step toward a theoretical conceptualization of coping with this stressor.

Study objectives: (1) To examine the association between the woman's cognitive perception regarding the lack of a child of the desired sex (henceforth, "perception of lack") at the time of the application to the Committee, and psychosocial adjustment measures (mental health and dyadic adjustment) some years later, at the time of data collection; (2) To examine whether dyadic coping at the time of application mediates the association between the perception of lack and psychosocial adjustment; (3) To examine whether coping resources (perceived self-efficacy, perceived social support) are directly related to and/or moderate the association between the perception of lack and psychosocial adjustment, and the association between the dyadic coping and psychosocial adjustment; (4) To examine whether the Committee's decision mediates the association between the perception of a lack and psychosocial adjustment; (5) To identify the variables which are the main contributors to psychosocial adjustment.

Methodology: The study design was cross-sectional and retrospective. The quantitative aspect of the research was preceded by a qualitative component, used for the construction of a questionnaire to measure the perception of lack.

Study population: The study population included 121 Jewish women who had applied to the Committee and had three or four children of one sex only. Most were Israeli-born, married, in their mid-thirties at the time of the application, worked full or part-time, and defined their economic status as good or higher. Only about one-fifth defined themselves as secular, with the remainder representing different levels of religiosity. Two-thirds of the participants had submitted an application to the Committee in the three years prior to the research interview; the others submitted applications between three and eight years before the research interview. Over one-half had applied for PGD in order to select a male child. The Committee's decision regarding the  participants' applications: 18 couples (14.8%) had their request approved;          24 couples (19.8%) had their request rejected; with the remaining 79 couples (65.4%) no decision was made by the time of the study, because the couple had not complete the required administrative process one year prior to the interview. In the years following their application to the Committee, about one-third of the participants gave birth to at least one more child, most to a child of the desired sex.

Data collection: The data were gathered from January 2016 to May 2017, after receiving approval from the Ministry of Health's Helsinki Committee. After completing and signing informed consent forms, participants were interviewed, in person (35%) or by telephone (64.5%), by means of a structured questionnaire.

Instruments: The application variables were the only variables extracted from the applicant's Committee file. All the other questionnaires were self-reported at the interview. These included: the background variables that served as control variables (demographics, fertility); four instruments that retrospectively examined variables related to the situation at the time of application to the Committee: (a) stress factor: a perception of lack questionnaire, constructed for this study; (b) coping styles: the Dyadic Coping Inventory (Bodenmann, 2007); (c) coping resources: the General Self-Efficacy Scale (Jerusalem & Schwarzer, 1992); and (d) two sub-indices of the MSPSS Perceived Social Support questionnaire (Zimet et al., 1988). Two questionnaires were used to examine psychosocial adjustment (the outcome variables) at the time of interview: the abbreviated Mental Health Inventory (Veit & Ware, 1983), and the Dyadic Adjustment Scale (DAS) (Spanier, 1976).

For a more in-depth understanding of the stressor, additional unique variables were examined at the time of interview. With the mothers who still did not have a child of the desired sex: her perception of lack over time; her current perception of lack; the extent of pressure regarding her children's sex from the environment in general, and the extent of pressure from specific sources. With the mothers who had since given birth to a child of the desired sex: her perception of the contribution that the child's birth had made to her self-perception, and social relationships.

Analysis: Bivariate associations and multivariate analyses to examine the mediation and moderation hypotheses were conducted using models of linear regression, logistic regression, and the Process procedure for mediation assessment.

Main findings: After adjusting for control variables, the multivariate analyses revealed a significant negative association between the perception of lack at the time of application to the Committee, and current mental health at the time of interview (β=-.31, p<.01). The greater the perception of lack, the lower the level of mental health. No association was found between the perception of lack at the time of the application and dyadic adjustment at the time of interview (β=-.08, p>.05).

An analysis of the mediation hypothesis revealed that dyadic coping only partially mediated the association between the perception of lack and mental health. Both direct (b=-.34, p<.05) and indirect associations through dyadic coping (b=-.10, p<.05) were found between the perception of lack and mental health, and a direct association was found between dyadic coping and mental health (b=.77, p<.01). In contrast, dyadic coping fully mediated the relationship between the perception of lack and dyadic adjustment (indirect =-.07, p <.05) while no direct effect was found (b=.02, p>.05). The greater the perception of lack, the lower the dyadic coping (b=-.13,p=.05); and the lower dyadic coping, the lower the dyadic adjustment (b=.51, p<.001).

An analysis of the moderation hypotheses indicated that they were not confirmed: coping resources did not moderate the relationship between the perception of lack and the psychosocial adjustment measures (mental health, dyadic adjustment), nor the relationship between the dyadic coping and adjustment measures. The only direct association found was that between "the support of a person who is close to me" i.e. the husband (a sub-index of the social support questionnaire) and dyadic adjustment.

An analysis of the association between the interviewees according to the Committee decision group (approved, rejected, or non-completion of the administrative processes) and explanatory and dependent variables did not provide any corroboration of the study's hypotheses. No association was found between decision group and the adjustment measures, but only a direct association with the perception of a lack. Further, it was found that the decision group variable did not mediate the relationship between the perception of lack and the adjustment indices. These findings should be addressed cautiously, given the small size of the decision groups. In examining the probability of belonging to a decision group, a positive correlation was found with the number of children, and a negative correlation with the woman's age at the time of the application to the Committee.

Regarding the contribution of the study variables to the explanation of psychosocial adjustment (mental health, dyadic adjustment), it was found that dyadic coping was the only variable that contributed to both adjustment measures. Regarding the other variables, perception of lack and (from the control variables) the number of children at the time of application contributed significantly to mental health. The support of a close person i.e., the husband, and (from the control variables) economic status, contributed significantly to dyadic adjustment. The remaining variables were not found to be contributors; nor were the desired child's sex, or whether the child of the desired sex was born after the application.

Conclusions, the research contribution and implications for research and practice: The findings of this study indicate, for the first time, that the lack of a child of a desired sex is an additional stress factor in the field of fertility. Moreover, the findings point to a direct association between this lack and the mother's consequent mental health, and an indirect association between this and consequent dyadic adjustment. The findings also highlight the centrality of the dyadic context in coping with the perception of lack.

These findings contribute to existing theories on coping with fertility problems in particular, and coping with stress in general. Beyond the study's contribution to advancing the understanding the psychosocial component of the perception of lack, it is possible to propose, for the first time, that the concept of infertility might be broadened to include "absent parenthood" as a form of "perceived infertility": a perception stemming from the woman's inner world, one that may manifest even if the woman doesn't suffer from fertility problems.

Another theoretical issue in the examination of this stressor, which requires further research, is the absence of a moderating effect by coping resources on the relationship between the perception of lack and psychosocial adjustment.

The findings of this study point to the importance and uniqueness of the psychosocial component in the Committee's overall considerations and its decisions, and support its policy that psychosocial assessment is a required stage of the application's administrative process. Although the findings are preliminary and deal with a unique population, they can nevertheless contribute to bioethical discussions on the subject, and can help set the framework for future policy regarding sex selection for non-medical reasons. It should be acknowledged, however, that this study has a number of limitations, mainly related to difficulties in accessing the study population, lack of representativeness, and its reliance on retrospective memory.

In view of the preliminary nature of the study, further research is required in order to confirm the results. For example, further research could focus on both partners of the dyad, various population groups, different stages of life, and the inclusion of various background and biopsychosocial variables. The Perception of Lack Questionnaire developed for this study may, after further validation, add to existing instruments a specific tool for examining this stress factor, and may contribute in the diagnostic and treatment processes involved in the Committee's decisions and in follow-up studies.

In terms of this study's practical contribution, it is important that professionals treating the mothers of children of only one sex are aware of the possibility of this stress factor, and assess its intensity and implications during diagnosis and care. This study and subsequent research can also contribute to theoretical and practical knowledge regarding the long-term consequences of coping with other fertility problems, cognitive perceptions of unique stress situations, parenting, and the bioethical issues that emerge through the development of medical technologies.