Priorities and Understanding of Pregnancy Among Women With Congenital Heart Disease

Background Many women with congenital heart disease (CHD) desire safe and successful pregnancies, but a significant proportion does not seek prepregnancy counseling. Objectives This study aims to distinguish the personal priorities and perceptions about pregnancy in this growing population. Methods Women aged 18 to 50 years with CHD were enrolled from 2 sites. Using a mixed-methods approach (Q-methodology), 179 participants sorted 23 statements representing a collection of views on pregnancy using priority forced ranking along a scale from “strongly agree” to “strongly disagree.” Results Majority of women were between 25 and 29 years of age, had moderate or severely complex CHD, and were married. Five unique group identities were elucidated from patient responses. Group 1 was centered around a strong desire to start a family. Women in group 2 had significant anxiety, and their psychological wellbeing interfered with their decision to start a family. Women in group 3 were concerned about premature death; if they do have kids, they want to be alive to see them grow old. Women in group 4 had strong objections to termination. Group 5 valued health care engagement. Group identities were unrelated to CHD complexity and demographic factors such as age and marital status. Six differentiating statements were identified that help distinguish which group a woman aligns with. Conclusions Women with CHD have diverse priorities and values relating to pregnancy and heart disease. This study used a mixed-methods approach to provide a framework identifying several domains for targeted prepregnancy counseling in women with CHD.

W omen represent more than one- half of the 1.4 million adults in the United States living with congenital heart disease (CHD), many of whom hope to experience safe and successful pregnancies. 1Historically, many women with moderate and severe CHD were advised against pregnancy.3][4][5] The increased survival and accelerated growth in the CHD population, improved quality of life, and rapid evolution in guidelines surrounding pregnancy have resulted in women receiving conflicting and often inaccurate guidance.7][8] The most recent 2018 American Heart Association/American College of Cardiology guidelines for managing adults with congenital heart disease (ACHD) emphasize the role of preconception counseling to avoid unplanned pregnancy in this population. 9rrently, prepregnancy counseling for women with CHD often focuses on maternal peripartum cardiovascular risks; however, this counseling has significant life, career, and relational implications.
Clinicians are rarely privy to these critical personal considerations because few receive specific training in pregnancy counseling. 10,11Understanding the personal and social values that influence a woman's decision (and invariably her health) to become pregnant can aid in providing tailored counseling to what each woman prioritizes and what is most likely to impact her long-term health, with the ultimate goal of providing individualized counseling, support, and avoidance of an unsupervised or unplanned pregnancy.
Our objective was to conduct a mixed-method research study, commonly utilized in psychology and social sciences, to assess individual "subjectivity" and to identify the spectrum of pregnancyrelated values from a representative sample of women with CHD. 12

METHODS
Women of childbearing age between 18 and 50 years, regardless of parity or gravity, with a diagnosis of CHD were prospectively enrolled using convenience sampling from May 2018 to October 2021 from multiple clinics at 2 large academic centers with comprehensive ACHD care (University of California-San Diego and Brigham and Women's/Boston Children's Hospital).After informed consent, demographic and clinical information was collected through surveys and the electronic medical record.CHD severity scores were assigned following the 2018 American Heart Association/American College of Cardiology guidelines for managing ACHD. 13 interview study guide was constructed after an extensive literature review.In-person unstructured interviews using this guide were conducted with key informants, and responses during these interviews served in piloting of the Q-sort and aided in refining the final draft.The Q-sort consisted of 23 statements representing a broad spectrum of views expressed around CHD and pregnancy (Table 1).Participants were required to sort all the 23 statements into a quasi-normal distribution of a 23-cell plot (Supplemental Figure 1) plotted along a numerical scale from À3 to þ3, corresponding to "Strongly Disagree" through "Strongly Agree", respectively.
The Q-sort was administered with physical cards for the first 52 patients, and the remainder was completed using the Q-Sort Methodology Software during clinic visits.Unstructured feedback was elicited from participants after completing the Q-sort exercise.
The Q-methodology uses priority forced ranking to identify participants who ranked statements similarly and calculates a factor loading score. 12,14The higher the factor loading score is, the more a participant identifies with a particular group (or factor group).
The analysis intentionally groups participants based on responses to the Q-sort and not by demographic data.
Statistical analysis was performed using the Q-method software, validated in other studies. 15Per previously published literature, 5 centroid factors were extracted, and 5 factors (referred to as groups from here on) were selected for varimax algorithm rotation. 14,15After rotational analysis, an individual participant's factor loading was considered significant at P < 0.05.A manual review followed this; participants loaded significantly onto more than 1 factor or did not load significantly on any factor were excluded.Weighted Z-scores were calculated to identify the distinguishing statements for each group.

RESULTS
A total of 179 women aged 18 to 50 years with CHD were enrolled.All participants who gave consent completed the Q-sort.More than three-quarters of participants were younger than 40 years, and the vast majority of participants had moderate or severely complex CHD and a history of cardiac surgery (Table 2).Two-thirds were married or in a serious relationship.Forty-two percent of women had been pregnant at least once, and 35% had given birth.
Approximately one-third of women who gave birth had a cesarean delivery, most of whom reported that their provider believed operative delivery was safer given their underlying heart disease (Table 3).Participant responses generated 5 unique group identities, which explained 56% of the cumulative variance.Interestingly, this group was the most likely to have received advice against pregnancy.One woman who identified with this group shared, "I consider myself fortunate and look forward to sharing information with my adult daughter who also has a congenital heart condition so she can make informed decisions."GROUP 2: PSYCHOLOGICAL WELLBEING IS NEGATIVELY IMPACTED BY CHD.Women who identify with this group have a good understanding of their CHD and previous interventions but overwhelmingly feel that CHD is a significant burden on their daily life and negatively affects their feelings of happiness to the point where they frequently worry about death.They do not feel strongly about having or carrying their own children.They feel strongly that physician approval is essential before attempting pregnancy, and if personal health is at risk, they would consider termination.The majority of women (75%) had never It is important that my partner wants kids þ2 À1 þ1 0 0 Herrick et al Mean number of pregnancies P ¼ 0.002 Herrick et al Understanding of Pregnancy in Women With CHD woman in this group shared, "I feel like women that have CHDs don't get the same answers from doctors when it comes to pregnancy."when trying to prevent pregnancy.Therefore, CHD is not a burden on their daily life, nor does it negatively affect their happiness.The majority had never been pregnant or given birth.A woman in group 5 shared, "I personally will not be having children.The risks involved sealed the deal when going over the current studies out there and the higher risk factors that would involve me being pregnant." GROUP AND SITE COMPARISONS.Age, marital status, race/ethnicity, and CHD complexity were not significantly associated with any group.were significantly more women who had a pregnancy and delivery in groups 1 and 4 than in the other groups.
There were no differences between the groups regarding planned vs unplanned pregnancy (P ¼ 0.12).In total, 38% of women reported they had been told at some point in their lives that they should not become pregnant, but none had a diagnosis with an absolute contraindication to pregnancy.
Of the 179 enrolled, 157 women loaded significantly with one of the 5 groups; the remaining 22 did not significantly load in any of the 5 groups (n ¼ 16) or Values are n (%).
Herrick et al Understanding of Pregnancy in Women With CHD loaded significantly into more than one group (n ¼ 6).
These results were validated using the Q-method software and a separate manual, iterative review.
There were no significant sociodemographic differences between women who did and did not fit into a specific group identity.
There were no significant differences between the 2 sites regarding age, CHD complexity, or group identification (Supplemental Table 1).However, site A had significantly more non-Caucasian patients, and site B had more patients who were married or in a committed relationship.Site-specific analysis yielded similar results to the combined analysis, except that site B analysis yielded no group identity driven by anxiety.
Six differentiating statements that quickly helped determine the group identity were identified (Table 4, Central Illustration).

DISCUSSION
With the growing population of women with CHD comes a growing need for specialized combined cardiovascular and obstetric care.This study is among the first to assess perspectives and experiences of women with CHD relating to pregnancy and childbearing, intending to streamline the process of providing individualized counseling on topics most relevant to each woman.Here we provide a categorization system within the heterogeneous patient population of women with CHD.
We  Herrick et al Understanding of Pregnancy in Women With CHD thorough explanation of risks and advanced care planning. 16This group felt neutral on having children, so they may benefit from a more-frank riskbenefit discussion on pregnancy to allow the individual to decide to maximize their overall health and longevity.Many women indicated they had either an unplanned pregnancy or no preconception counseling before a planned pregnancy (33.8% and 32.4%, respectively).Interestingly, there were no betweengroup differences (P ¼ 0.115) in preconception counseling, highlighting the need to make pregnancy planning a routine part of ACHD care for all patients, regardless of their group identity.Women currently in their 30s and 40s have seen a dynamic shift in ACHD and pregnancy guidelines throughout their lifetime.In fact, 38% of women reported they had received advice not to become pregnant although, by current guidelines, none had a diagnosis consistent with an absolute contraindication to pregnancy.
Receiving conflicting advice makes it difficult to decide which to follow and may lead to avoidance of care, as seen by the relatively high number of women who reported having a planned pregnancy but did not meet with a physician prior.Additionally, one-third of women had unplanned pregnancies, which speaks to the importance of contraception counseling as part of routine ACHD care.Despite the advice to avoid pregnancy, 27 of the 68 women became pregnant.
We observed between-site differences in anxiety and the burden of CHD on daily life.Most patients enrolled from site B were in the pediatric setting, which may have impacted the patient's understanding of CHD, self-care, and subsequent anxiety and CHD as a burden on daily life.The subtle differences in group identities between the 2 sites further highlight the potential benefits of using the distinguishing statements as a tool to enhance patient-centered care. The

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Categorical variables were summarized with frequencies and percentages, and continuous variables with median and standard deviation unless otherwise indicated.Summary statistics were calculated in SPSS (version 26, IBM).The study was approved by the Institutional Review Board of the University of California San A B B R E V I A T I O N S A N D A C R O N Y M S ACHD = adults with congenital heart disease CHD = congenital heart disease Herrick et al J A C C : A D V A N C E S , V O L . 1 , N O . 4 , 2 0 2 Understanding of Pregnancy in Women With CHD O C T O B E R 2 0 2 2 : 1 0 0 1 1 2 Diego, Boston Children's Hospital, and Brigham and Women's Hospital.The study procedures complied with Health Insurance Portability and Accountability Act regulations and obeyed the tenets put forth by the Declaration of Helsinki.

GROUP 1 :
A STRONG DESIRE TO START A FAMILY DESPITE CHD.Women who identified in this group felt strongly about carrying a child themselves (less likely to consider adoption), finding a partner who also wants children, and being alive to see their children get married.The defining statement for this group is, "My life would be incomplete without children."They feel neutral about physician approval before conception.They exhibited no significant anxiety related to their CHD, nor was it a burden on daily life.Approximately one-half of the participants have been previously pregnant and given birth.

GROUP 4 :
STRONG OBJECTION TO TERMINATION OFPREGNANCY.Women who identify with this group would not consider termination regardless of gestational age, even if their health was at risk.They feel neutral to slightly positive about having and carrying their children and endorse having a good understanding of the cardiovascular risks of pregnancy.Previous pregnancy and birth strongly distinguish this group from others.However, only one-third who identified with this group and had given birth received preconception counseling.A woman in this group shared, "This is a great study that I hope provides comfort for women to make a better plan for their future whether they decide to have children or not.I would press that the overall health is taken into account."GROUP 5: NEED FOR EMPOWERMENT AND CONTINUOUS HEALTH CARE ENGAGEMENT.Women who identify with this group endorse an in-depth understanding of their CHD.Through continuous health care engagement, they know what procedures have been done to their heart and the health risks associated with pregnancy.They accentuate the importance of physician approval before attempting pregnancy and emphasize the role of birth control

Groups 4
and 5 both endorsed a high-level understanding of their CHD and partner involvement; however, they diverged on the distinguishing statements.Group 4 was distinguished by a strong objection to termination, and group 5 focused on high health care engagement.While there are similarities CENTRAL ILLUSTRATION Group Identities Distinguishing Statements and Recommended Counselling Herrick N, et al.JACC Adv.2022;1(4):100112.J A C C : A D V A N C E S , V O L . 1 , N O . 4 Understanding of Pregnancy in Women With CHD between the groups, viewed from a clinical perspective, focusing on the differentiating factors may be useful when devising a counselling strategy.For example, if a woman who identified with group 4 desires pregnancy, it would be critical to provide indepth preconception counselling (including clear guidelines on timing and specifics of cardiac optimization) as well as inquiry about the rationale behind the objection to termination (cultural, faith-based, misconception, personal, etc).Thus, emphasizing the distinguishing statements is more likely to yield more effective and open-ended inquiry and adequate counseling.

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Herrick et alJ A C C : A D V A N C E S , V O L . 1 , N O . 4 , 2 0 Understanding of Pregnancy in Women With CHD O C T O B E R 2 0 2 2 : 1 0 0 1 1 2 pregnancy and childbearing, which can help inform individual clinicians as well as public health interventions to improve the experiences of women with CHD that consider becoming pregnant.Traditional demographic factors, including age and marital status, were not predictive of opinions relating to pregnancy.Six of the 23 statements were identified as distinguishing; the answers can help identify which group identity a woman aligns most closely with, free of demographic bias, and provide a framework for clinicians to identify efficiently, in an emotionally sensitive manner, what an individual woman prioritizes to determine appropriate timing for referral to maternal-fetal medicine and improve patientcentered care.ACKNOWLEDGMENTS The authors want to express their gratitude to all the women who participated in the study.The survey questions invoked emotional responses for most-the authors are grateful that all who started the Q-sort finished it to completion.The authors feel inspired to be working with this special population, all of whom expressed the desire to help the next generation of women living with congenital heart disease.

TABLE 1 Q
-Sort List of Statements

TABLE 3
Details of Gravity, Parity, Preconception Counseling, and Delivery Recommendations by Group Identification

TABLE 4
Distinguishing Statements for Each of the Factor Groups and Sample Survey to Help With Rapid Identification of Group Type Dr Alshawabkeh is supported by the American Heart Association Career Development Award grant.Dr Al-Rousan is supported through a grant from the National Heart, Lung and Blood Institute (#K23HL148530).Ms Lee, Dr Rodriguez, and Dr Valente are supported by the Sarah Marie Liamos Fund for Adult Congenital Heart Disease Research.All other authors have reported that they have no relationships relevant to the contents of this paper to disclose.