What Do Men Want? Exploring Gender Differences in Supportive Care Preferences of Couples With Recurrent Pregnancy Loss


 Background International guidelines recommend to offer supportive care during a next pregnancy to couples affected by recurrent pregnancy loss (RPL). In previous research, several options for supportive care have been identified and women’s preferences have been quantified. Although it is known that RPL impacts the mental health of both partners, male preferences for supportive care have hardly been explored. Methods A cross-sectional study was conducted in couples who visited a specialized RPL clinic in the Netherlands between November 2018 and December 2019. Both members of the couples received a questionnaire that quantified their preferences for supportive care in a next pregnancy and they were asked to complete this independently from each other. Preferences for each supportive care option were analyzed on a group level (by gender) and on a couple level, by comparing preferences of both partners. Results Ninety-two questionnaires (completed by 46 couples) were analyzed. The overall need for supportive care indicated on a scale from 1-10 was 6.8 for men and 7.9 for women (P = 0.002). Both genders preferred to regularly see the same doctor with knowledge of their obstetric history, to make a plan for the first trimester and to have frequent ultrasound examinations. A lower proportion of men preferred a doctor that shows understanding (80% of men vs. 100% of women, P = 0.004) and informs on wellbeing (72% vs. 100%, P = ≤0.000). Fewer men preferred support from friends (48% vs. 74%, P = 0.017). Thirty-seven percent of men requested more involvement of the male partner at the outpatient clinic, compared to 70% of women (P = 0.007). In 28% of couples, one partner desired peer support, while the other partner did not prefer this.Conclusions While both women and men affected by RPL are in need of supportive care, their preferences may differ. Current supportive care services may not entirely address the needs of men. Health care professionals should focus on both partners and development of novel supportive care programs with specific attention for men should be considered.


Abstract Background
International guidelines recommend to offer supportive care during a next pregnancy to couples affected by recurrent pregnancy loss (RPL). In previous research, several options for supportive care have been identi ed and women's preferences have been quanti ed. Although it is known that RPL impacts the mental health of both partners, male preferences for supportive care have hardly been explored.

Methods
A cross-sectional study was conducted in couples who visited a specialized RPL clinic in the Netherlands between November 2018 and December 2019. Both members of the couples received a questionnaire that quanti ed their preferences for supportive care in a next pregnancy and they were asked to complete this independently from each other. Preferences for each supportive care option were analyzed on a group level (by gender) and on a couple level, by comparing preferences of both partners.

Results
Ninety-two questionnaires (completed by 46 couples) were analyzed. The overall need for supportive care indicated on a scale from 1-10 was 6.8 for men and 7.9 for women (P = 0.002). Both genders preferred to regularly see the same doctor with knowledge of their obstetric history, to make a plan for the rst trimester and to have frequent ultrasound examinations. A lower proportion of men preferred a doctor that shows understanding (80% of men vs. 100% of women, P = 0.004) and informs on wellbeing (72% vs. 100%, P = ≤0.000). Fewer men preferred support from friends (48% vs. 74%, P = 0.017). Thirty-seven percent of men requested more involvement of the male partner at the outpatient clinic, compared to 70% of women (P = 0.007). In 28% of couples, one partner desired peer support, while the other partner did not prefer this.

Conclusions
While both women and men affected by RPL are in need of supportive care, their preferences may differ.
Current supportive care services may not entirely address the needs of men. Health care professionals should focus on both partners and development of novel supportive care programs with speci c attention for men should be considered.

Background
Recurrent pregnancy loss (RPL) is a frustrating condition for both patients and care providers. This condition, de ned as the loss of two or more pregnancies before the fetus reaches viability, is estimated to affect 1-3% of all couples of reproductive age. [1][2][3] Multiple risk factors have been identi ed, but despite extensive diagnostic investigations, RPL remains unexplained in the 60-70% of cases. [4] For these couples, there is currently no evidence-based medical treatment option. As pregnancy losses are generally experienced as signi cant negative life events, RPL may have serious psychological impact. A recent study reported that both women and men affected by RPL show high risks for developing depression and anxiety, while they often use different coping strategies. [5] It is recommended by current international guidelines to offer supportive care programs for couples with RPL. [6] Some studies even suggested that supportive care during early pregnancy may have a bene cial effect on pregnancy outcome, although this evidence is limited. [7][8][9][10] Moreover, professional support and compassionate care are highly valued by couples with RPL. [11] Musters et al. elucidated what is actually perceived as supportive care for RPL and evaluated women's preferences for twenty supportive care options during a next pregnancy. [12,13] They showed that women with RPL preferred to see the same doctor during their consultations who is specialized in RPL, takes them seriously, listens, shows understanding and enquires about emotional needs. The women wanted to make a plan with their doctor for the rst trimester of a new pregnancy and they preferred frequent ultrasound examinations during this period. Furthermore, they indicated a need for psychological after-care in case of a new miscarriage.
Notably, male partners' preferences and their need for supportive care were not addressed in this study.
That partner inclusion has been omitted in most studies in this eld, is shown by a systematic review [14] that evaluated 27 studies on patient-centered early pregnancy care. The male perspective was examined in only three of the included studies and the authors considered involvement of the partner as an improvement target. Identifying male preferences for supportive care in RPL is relevant, not only because it has been shown that men do also suffer from RPL, but also because tailored supportive care programs may assist the male partner during a new pregnancy. The signi cance of this has been underscored by several studies showing that the male role in pregnancy is of great impact on maternal health behavior and pregnancy outcome. [15][16][17] The aim of the current study was to quantify preferences for supportive care of both men and women affected by RPL. Previously identi ed supportive care options for RPL [12,13] were used as a framework for this study and both members of participating couples were independently questioned, allowing us to compare preferences between genders but also to analyze potential discrepant preferences within couples.

Participants
The study was conducted in couples that visited the specialized RPL outpatient clinic of the Leiden University Medical Center in the Netherlands between November 2018 and December 2019. Couples had at least two pregnancy losses (following the de nition of the ESHRE guideline for RPL [1]) and were uent in Dutch or English. The study protocol was approved by the Medical Research Ethics Committee of the Leiden University Medical Center (reference number N19.101). All participants provided written consent to take part in the study.
Procedures at the RPL outpatient clinic When couples visit the RPL clinic for the rst time, they have an intake consultation with a gynecologist or fertility doctor. The team comprises four physicians, all specialized in RPL. All physicians adhere to the same protocol and provide similar care. New patients are discussed in the team after their rst consultation. Besides obtainment of detailed obstetric history and extensive history of both partners, couples receive information about known risk factors for RPL, advices on lifestyle changes, options for diagnostic testing, potential therapeutic options, chances for future pregnancy outcome and ongoing studies.
Options for diagnostic investigations include screening for uterine anomalies (transvaginal 3D ultrasound), thyroid abnormalities (anti-thyroid peroxidase (TPO) and thyroid-stimulating hormone (TSH) levels), parental karyotyping (only in case of increased risk of abnormalities, according to the risk table of Franssen et al. [18]) and acquired thrombophilia (antiphospholipid syndrome (APS)). Screenings for inherited thrombophilia and sperm factors (DNA fragmentation and seminal plasma cytokine levels) are only carried out for research purposes. [19,20] Therapeutic options are discussed in case of a diagnosed risk factor associated with RPL, following recommendations of the ESHRE guideline. [1] Besides the medical approach, attention is paid to the psychological impact of RPL and consultation with a medical social worker is offered. In case of a next pregnancy, couples are offered monitoring at the RPL outpatient clinic in the rst 12 weeks of the pregnancy. Ultrasound examination in the rst trimester is offered, the frequency depending on the couple's preference. In addition, it is emphasized that the clinic is available 'twenty-four seven' and can be reached in case of any symptoms or distress. In case of an ongoing pregnancy beyond 12 weeks, the couple will be referred for further regular monitoring of the pregnancy to either an obstetrical outpatient clinic or a midwifery practice (depending on medical indication and individual situation). In case of another pregnancy loss, the doctor will re-evaluate their individual plan at the follow-up consult at the RPL outpatient clinic.

Data collection
The questionnaire consisted of two parts: general demographic questions and preferences for supportive care. The second part of the questionnaire was based on supportive care options in three domains as identi ed by Musters et al. [12,13]: 1: Medical supportive care (for example: ultrasound examination during early pregnancy, medical information and advices); 2: Soft-skills (for example: communication skills of the doctor) and 3: Other types of supportive care (for example: support from friends, family and peers, relaxation exercises, alternative therapies).
Two versions of the questionnaire were used, intended for either women or men. Given the purpose of the study, the couples were asked to complete the questionnaires independently, without discussion between both partners. The questionnaires were available in Dutch and English language (the English version is included as Supplementary material). Preferences and need for supportive care were quanti ed using 5point Likert scale items ranging from total disagreement to total agreement and a rating scale question (grade 1-10). The estimated completion time for the questionnaire was maximum 15 minutes. The questionnaires were developed and pilot tested by two gynecologists (specialized in RPL), two fertility doctors (specialized in RPL), a psychologist, a PhD candidate (specialized in RPL) and two patients with RPL. No major adjustments were made after pilot testing.

Statistical analyses
Descriptive data are presented in numbers and percentages. The 5-point Likert scale items for supportive care options were recoded: 1 and 2 represent the non-preference group, 3 the neutral group, and 4 and 5 the preference group (similar to Musters et al. [13]). To prevent multiple hypothesis testing, statistical tests were not executed for the complete panel of supportive care options but restricted to prede ned selected entities: whenever a supportive care option was preferred by either ≥60% of women, ≥60% of men, or both, this option was considered as potentially relevant for clinical practice and thus examined in further detail. This was done by comparing the preference rates for these selected supportive care options between women and men with a McNemar test for paired categorical data. The mean overall need for supportive care expressed on a scale from 1-10 is presented with standard deviation (SD) and compared between women and men with a paired samples T-test. Two-sided P-values <0.05 were considered statistically signi cant. Intra-couple discrepancy was de ned as one of the two partners having no need (1 or 2) for a certain supportive care option and the other partner having a preference (4 or 5) for this supportive care option. The level of intra-couple discrepancy for each supportive care option was calculated as the percentage of all couples that met this de nition. Analyses were performed in R studio version 1.3.9.50 (R Foundation for Statistical Computing, Vienna, Austria).

Sample size calculation
On the basis of the null hypothesis that an equal percentage of women and men would prefer a supportive care option, a sample size of 44 couples would be required for an 80% power at a two-sided alpha of 0.05 to detect a difference in preference rate of 30% between women and men, which we considered as a clinically relevant difference. The sample size was calculated with R studio package 'SampleSizeMcNemar'.

Results
Between November 2018 and December 2019, 50 women and 46 men completed the questionnaire. Four questionnaires were excluded from the analyses as only the female partner returned the questionnaire. All couples were heterosexual. The majority of women and men (85% both) was born in the Netherlands. The median number of pregnancy losses at the time the RPL outpatient clinic was visited for the rst time was 2 (range 2-6). No underlying condition for RPL was found in 70% of the couples. More baseline characteristics of the couples are shown in Table 1.  Preferences for supportive care in a next pregnancy The mean need for supportive care expressed on a scale from 1-10 was 6.8 (SD 1.68) for men and 7.9 (SD 1.65) for women (P = 0.002). Seventeen options for supportive care in a next pregnancy were preferred by either the majority (≥60%) of women and/or men. Preference rates and levels of intra-couple discrepancy for these speci c options are shown in Figure 1, including P-values for the differences in preference rates between women and men. An overview of the other supportive care options, being preferred by <60% of women and men, is shown in Figure 2.

Domain 1: Medical supportive care
The majority of both women and men preferred making a plan for the rst trimester, seeing the same doctor during different consultations who has knowledge of their obstetric history, an ultrasound examination directly after a positive test, once a week during the rst trimester and during symptoms and medication for RPL that is proven safe for pregnancy. Medication that is not proven safe during pregnancy ('medication in general') was preferred by 33% of women and 24% of men. Information derived from a doctor was preferred over information derived from the internet or information derived from peers. On group level, there were no signi cant differences between genders for all of the above options. The levels of intra-couple discrepancy were highest for the options information from peers (26%), information from the internet (24%) and advice regarding lifestyle (22%).

Domain 2: Soft skills
The majority of men and women preferred a doctor that takes the patient seriously, listens, informs on emotional needs, shows understanding and informs on wellbeing. For the last two options the preference rates signi cantly differed between women and men. Showing understanding was preferred by 100% of women vs. 80% of men (P = 0.004). Informing on wellbeing was preferred by 100% of women vs. 72% of men (P = ≤0.000). Couples had most discrepant preferences towards counselling from a specialized nurse (level of intra-couple discrepancy 17%; preferred by 52% of both women and men) and counselling from a psychologist (level of intra-couple discrepancy 17%; preferred by 24% of women and 13% of men).

Domain 3: Other types of supportive care
Options being preferred by the majority of women were: support from friends, support from family, more involvement of the male partner at the outpatient clinic (i.e. the doctor actively involves the male partner during consultations and in supportive care) and to talk to someone after a new miscarriage. The proportion of men that expressed a need for support from friends was signi cantly lower (48% vs. 74%, P = 0.017). None of the options in this domain were requested by ≥60% of men. More involvement of the male partner at the outpatient clinic was preferred by 70% of the women, compared to 37% of the men (P = 0.007). Sixty-one percent of women would like to talk to someone after experiencing another miscarriage, compared to 43% of men. The highest levels of intra-couple discrepancy were observed for need for support from peers (28%), followed by relaxation exercises (24%), yoga (24%) and talking to someone after a new miscarriage (22%).
Overall, the options for supportive care that were rejected by the majority of both women and men were bereavement therapy, listening to relaxation tapes, counselling from a social worker, counselling from a psychologist, alternative medication and hospital admission at the same gestational age as earlier miscarriages occurred. Alternative therapy (such as acupuncture or re exology), relation exercises and yoga were not considered necessary by the majority of men.

Discussion
This is the rst study that quanti ed preferences for supportive care of both men and women affected by RPL and explored the existence of different needs within couples. Overall, men expressed a signi cantly lower need for supportive care compared to women. Regarding medical supportive care, preferences of both genders were largely similar and in line with the previous study in women by Musters et al. [13]. For the other domains of supportive care, several between-gender differences were observed.
Although the majority of both men and women preferred a doctor that takes the patient seriously, listens, informs on emotional needs, informs on wellbeing and shows understanding, a signi cantly smaller proportion of men appreciated the last two options (differences of 28% and 22% compared to women, respectively). In addition, the majority of women expressed a need for support from family, friends and peers; men preferred this less. This is in accordance with previous research showing that men are typically more hesitant to disclose their feelings after pregnancy loss. [5,21] Although men do experience feelings of grief, stress and vulnerability, these emotions may be less manifested. [22,23] Men are thought to employ different coping strategies compared to women, including 'active avoidance' and distractive behavior, related to more frequently observed risk behaviors such as excessive alcohol consumption and smoking. [5,21] Multiple studies showed that a signi cant part of men affected by pregnancy loss experienced little support from their social network and a reluctance to share their loss and feelings with them; their family and friends tend to direct their acknowledgement and support largely toward the female partner. [5,24,25] Also in hospital settings where support activities are profoundly targeted on or delivered by women, men have indicated that they feel excluded or marginalized from care compared to their partner. [26] In our study, remarkable gender differences were observed regarding the overall need for supportive care (mean grade 6.8 in men vs. 7.9 in women) and the need for more involvement of the male partner at the RPL outpatient clinic (desired by 37% of men and 70% of women). This seems in contrast with other studies indicating that male partners of RPL couples want to be more included. [11,14] Multiple explanations may be underlying here. In some men's responses, a social desirability bias may be present. Various studies on experiences following pregnancy loss showed that it is not uncommon for men to view their role as primarily being a 'supporter' to their female partner, leading to a barrier to seek support for themselves. [21,[27][28][29] Another possibility is that the approach at the clinic and the supportive care as it is currently being offered, do not completely meet the needs of men.
Furthermore, our results suggest that it is important to offer supportive care services to both partners individually. Although men and women may show similar preferences on group level, this does not automatically imply a high level of intra-couple agreement. For instance, while an equal percentage of the total groups of women and men (52%) preferred counselling from a specialized nurse during a next pregnancy, in almost one in ve couples the partners had opposing opinions regarding this aspect (level of intra-couple discrepancy 17%). Moreover, in 28% of couples, one partner expressed a need for peersupport, while the other partner did not consider this necessary.
The major strength of this study is that it is the rst that quanti ed the need for different aspects of supportive care of both men and women affected by RPL. In a recent exploratory study in 13 couples with RPL, both members of the couples were interviewed simultaneously on their need for treatment, support and follow-up. [11] This likely resulted in each partner in uencing the other's perspectives, which was also recognized as a limitation by the authors themselves. In our study, the questionnaires returned by both members of each couple were carefully compared and no obvious overlap in their responses was present.
This makes it credible that the questionnaires were completed independently of one another (as requested), although we cannot entirely rule out the possibility of some couples having discussed their responses. The study has several limitations. First, it is a single center study and although the sample is representative for our RPL clinic, differences with RPL couples elsewhere may exist, for instance in terms of education level, being relatively high in our population. Second, the panel of supportive care options evaluated in this study was based on previous research restricted to women. It may be that some men desire other possibilities for supportive care, not being covered in this study.
It should be considered to develop supportive care programs for RPL speci cally aimed at men, as supportive care in its current form may not entirely suit their needs. In a previous qualitative study, men affected by (single) pregnancy loss expressed a desire for informally catching up with another man with the same experience. In a hospital setting, they suggested the option of a male support worker. Such possibilities may be further explored for men affected by RPL, for instance using focus group research.

Conclusions
Our study shows the existence of different preferences for supportive care of men and women affected by RPL. It is important that health care providers are aware of this and take a tailored approach. We recommend to actively involve both partners, ask them about their personal preferences and discuss the most suitable approach that best ts the needs of both partners. It can be emphasized that some supportive care services may be chosen by one of the partners only. In addition, development of maleoriented supportive care programs should be explored. Availability of data and materials The datasets generated and/or analyzed during the current study are available from the corresponding author on reasonable request.