Sampling and recruitment

This is a prospective qualitative analysis that examined the experiences of birthing parents who had a fetus with severe CHD and either terminated their pregnancy or elected for a hospice birth plan at Ann & Robert H. Lurie Children’s Hospital of Chicago (hereafter, “Lurie Children’s”) or Northwestern University Prentice Women’s Hospital. Severe CHD is defined as defects that require neonatal intervention for survival (e.g., single ventricle). Eligibility was determined through purposive sampling of birthing parents who had received multidisciplinary counseling for severe fetal CHD between 6/1/2019 and 3/31/2023 at Lurie Children’s. We did not recruit patients who received multidisciplinary counseling before 2019 to avoid recall bias. We chose March 2023 as the upper bound to allow at least nine months of bereavement time, between receipt of multidisciplinary counseling and study recruitment [21,22].

This purposive sample includes individuals who received counseling before and after the 2022 Dobbs v. Jackson Women’s Health Organization (Dobbs) ruling, which revoked the constitutional right to termination and returned authority to regulate termination to individual states, thus impacting access to care in certain geographic areas of the United States [23,24]. In Illinois, the Illinois Reproductive Health Act, signed in 2019, was strengthened after Dobbs, making Illinois a destination for out-of-state patients seeking termination [25].

Multidisciplinary counseling at Lurie Children’s entails a combined meeting with specialists from fetal cardiology, genetics, maternal-fetal medicine, neonatology, perinatal palliative care, pediatric cardiac intensive care, pediatric cardiac surgery, and social work, typically within four weeks of receiving a diagnosis of severe fetal CHD. Not all individuals who receive multidisciplinary counseling are offered a hospice birth plan; single ventricle physiology would be offered, while biventricular physiology (e.g., transposition of the great arteries) would not. Individuals who elected for postnatal surgical intervention, or who did not receive multidisciplinary counseling at this academic center, were ineligible for recruitment. All individuals who received multidisciplinary counseling had previously received initial counseling from a fetal cardiologist, social worker, and fetal nurse coordinator prior to their multidisciplinary counseling session, in accordance with standard practice [7]. Counseling providers varied between multidisciplinary counseling sessions, except for social work and palliative care, for which the same individuals were present for each session.

Eligible participants were contacted by email and telephone at least three times about the study and provided written or verbal consent prior to participation. All participants completed a questionnaire pertaining to demographic data prior to their interview; no electronic health record data were used in analysis. All participants were offered a $100 gift card for their participation. This compensation value was determined based on standard practice for qualitative interview research on sensitive topics at our institution.

Interviews

The interview guide (Supplemental Document 1) contained 22 open-ended questions that addressed the prenatal counseling experience, decision making about management, and for those who underwent a hospice birth plan, the postnatal hospice experience. The interview guide was created with collaborative input from fetal cardiologists (JW, SP, AP), neonatologists (JTF, NH), palliative care specialists (GK, JTF, NH), maternal-fetal medicine specialists (WG, LY), a bioethicist (AP), and two parents from the purposive sample.

An a priori target recruitment of 10 interviews was established by the author team, unless saturation was not achieved with this goal. Saturation, defined by the themes identified by the interviewer following their own review of interview transcripts prior to coding, was achieved after 7 interviews. However, given that primary and secondary coding of the transcripts would be conducted by three different analysts (and not the interviewer), an additional three interviews were conducted in case that additional themes might be identified. The recruitment period was September 1, 2023 to March 30, 2024. Patient interviews were conducted between October 2023 and April 2024.

Recruited participants underwent individual virtual interviews with a trained research team member (GK). The interviewer is an advanced nurse practitioner specializing in perinatal palliative care who was present at each participant’s multidisciplinary meeting and thus had existing rapport and experience with discussing highly sensitive topics with an empathic, nonjudgemental, and open-ended style. Each interview lasted 60–90 minutes. The lack of blinding between interviewer and participants was to foster openness given the complexity and sensitive nature of the subject matter, though social desirability in patient responses was possible. The interviewer intentionally adhered closely to the interview guide to mitigate bias and ensure consistency/dependability across interviews. The interviewer identifies as a cisgender, non-minority woman without lived experience of the topic. Although involved in patient care, she did not have authority over neonatal disposition after birth. All interviews were recorded, professionally transcribed, and deidentified prior to analysis.

Analysis and ethical oversight

Thematic analysis was conducted in five stages, using a combined deductive-inductive approach. First, a deductive codebook was created by three team members (DM, LB, JW) who identified initial codes based on the interview guide. Next, comparison coding was conducted based on three randomly chosen interviews, and the codebook was iteratively refined based on discussion of comparison coding (DM, LB, JW). Third, primary and secondary coding of each of the 10 interview transcripts were performed, with additional refinement of the codebook, and an initial narrative report outlining codes was written (DM, LB). Fourth, four team members independently reviewed the narrative report, and arranged the content into themes (GK, HS, JW, LY). Finally, to enhance credibility and confirmability, remaining team members (JTF, WAG, NH, AP, SP) addressed reflexivity with refinement and re-aggregation of themes.

Analysis was conducted using Dedoose (Version 9.0.46, 2021. Los Angeles, CA: SocioCultural Research Consultants, LLC). To protect participant privacy, limited demographic and clinical descriptive data are presented. To further ensure confirmability and authenticity, each subtheme was described with a direct interview quotation from three separate participants – one within the text, and two within the tables. The analysis is presented in accordance with the SRQR reporting guidelines (Supplemental Document 2). The study was approved by the Institutional Review Board at Lurie Children’s (2023–6213).

Barriers to decision making related to prenatal counseling

One theme, areas for improvement in counseling, was dominant. Five subthemes were identified: insufficient discussion about either termination or hospice birth, perceived lack of urgency, inconsiderate behaviors, lack of acknowledgment of impact of fetal CHD on family unit, and insufficiently specific or transparent language. Regarding insufficient discussion, one participant noted: “I had to… ask for that [surgical option for termination] and really push for what does that actually mean.” Others perceived a lack of urgency regarding access to multidisciplinary counseling: “I wish they [multidisciplinary counseling] would be more easily accessible that you can talk right away to a doctor that would do a surgery.” Some participants noted inconsiderate behaviors from counseling providers: “[counseling provider] gave us a newborn onesie that said, like little heart warrior… I found that offensive and distasteful...”

Many participants noted areas for improvement in counseling content, such as the lack of acknowledgement of how other family members may be affected: “Before making that decision [termination], there was no real mention of how, from what I remember anyway, her [sibling’s] life could potentially be impacted.” Another content-related subtheme was the desire for more specific or transparent language: “After going through a surgical D&E termination, I do feel like my understanding of what [the specific process of termination] was going to happen was obviously different than actually what happened…”

Facilitators to decision making related to prenatal counseling

The dominant facilitator was perceived strengths of counseling practices. Six subthemes were identified: value of multidisciplinary counseling, transparency about outcomes, respect for patient autonomy, patience and understanding, nonverbal communication practices, and use of written materials. Many participants noted the value of multidisciplinary counseling: “…there was 10 different people… And then if there was questions that somebody didn’t know, somebody else would jump in, and that was helpful.”

Participants also appreciated transparency about outcomes: “I needed the facts to be able to understand, I guess, how serious it [the diagnosis] was.” Participants appreciated language that respected their autonomy: “if somebody leaned a certain way, I think it maybe would have given us false hope.” Some participants noted the importance of nonverbal communication practices, for instance, the physical location and setting for counseling and particularly appreciated written tools: “…and [the cardiologist] drawing not just this is what the current state is, but what would happen if we had the surgery and that helped me a lot.”

Context-dependent facilitators or barriers to decision making related to prenatal counseling

Two themes, patient attributes specifically affecting counseling and views on provider recommendation contained subthemes that could act as either barriers or facilitators, depending on individual context and preferences. Barrier-focused subthemes included shock and information overload, previous knowledge or attitudes toward termination or hospice, and undesired bias. Most participants noted that they were initially shocked and overwhelmed after receiving a diagnosis of fetal CHD, which was a barrier to processing information: “I don’t know if I’ve repressed or suppressed some of these memories...” Several participants noted that their previous knowledge or attitudes toward termination served as a barrier to their decision. A participant also described their previous understanding of hospice as a barrier to their decision: “I think my immediate reaction to it [hospice] was that it was so passive, and just almost non-committal.” Finally, some participants described undesired bias against their final decision to pursue hospice birth: “I could have ended up delivering with her [midwife who was not supportive of decision] and that would have been very hard.”

However, in other contexts, these subthemes emerged as facilitators, including previous knowledge or attitudes toward hospice birth, and desire for provider recommendation. For some participants who chose hospice birth, previous knowledge about hospice was a facilitator to hospice birth counseling: “I had a couple of family members that had been on hospice... I knew what hospice was and what it entailed and everything.” Finally, although most participants described a preference for, and experience of, neutral counseling, a few participants expressed a desire for provider recommendation: “sometimes I wish that there was more of a bias because I felt like it would make me feel like I didn’t have to make this decision.”

Domain 2: Individual factors affecting decision making (Table 2)

Six themes emerged regarding individual considerations in decision making: emotions affecting decision making following counseling, value for reproductive autonomy (i.e., the ability to make informed decisions about reproduction free from coercion or constraint) [26], family factors, internal factors, outside input, and desire for resources. First, regarding emotions impacting decision making, the subthemes of guilt or uncertainty were common. One participant described guilt in the context of their living child: “of course I would do whatever for her [living daughter], so then it tore me to be like, well, I’m not going to do whatever for him [fetus with CHD]…” Another participant explained how uncertainty was driven by institutional counseling practices: “…after my initial consultation at [Institution 1]…”Oh, it was just a couple of surgeries and we can be all good.” I think after [consultation at Institution 2] is when I was like, okay…it’s not good…” Many participants who experienced termination described a fear of stigma for their decision, but those who chose hospice experienced stigma also: “our neighbor…was not very supportive. And I didn’t go into details with everybody...”

Regarding the theme of value for reproductive autonomy, analysis revealed subthemes of reproductive justice and shifts in opinion about termination. Specifically, reproductive justice refers to the emphasis on the human right to maintain bodily autonomy, have children or not have children [27]. With regards to reproductive justice, a participant who chose hospice still described her appreciation for having the option of termination: “even though I didn’t do it [termination]… it was still an option that I could have chosen...” Another participant described opinion shifts in a close family member due to their experience: “This experience of mine has really completely changed his [family member’s] perspective of that [termination]… he was very clear from the get-go like that I should make the choice that’s right for me.”

Regarding the theme of family factors that affect decision making, six subthemes were identified: spousal disagreement, baby’s suffering, baby’s long-term outcomes, effects on living children, financial consequences, and insurance and access to care. Regardless of whether a participant experienced termination or hospice, there were cases of spousal disagreement. In these cases, the birthing parent was the final decision maker, as one participant who elected for hospice birth described: “My husband was leaning more towards the termination route. But my body, so I told him like, I’m not doing that.” One participant also summarized their desire for information on long-term outcomes: “…about their IQ level, like, how much is it going to go down? Is he going to be able to do certain things?”

Several participants discussed the effects of caring for a child with CHD on other living children: “We were not sure how we were going to take care of one while the other one needed some help…. it’s hard to find help with the kids even if you can afford to pay.” Many participants described financial consequences of having a child with a chronic disease: “…then if you need help, how will insurance cover it [healthcare costs], we’ll be paying out of pocket. I think monetary, I mean, we’re all concerned about that I think.” Another participant described insurance as a major consideration in decision making: “I’m the one that has our insurance…so then if I wasn’t working, where would our insurance come from?”

Many reflected about internal factors which affected their decision making. These subthemes included personal risk tolerance, religious beliefs, and shifts in personal priorities. One participant described their own limited risk tolerance for poor outcomes: “…just knowing that there were those super negative options, that was enough…” Another participant who elected for hospice birth described how their religious beliefs shaped their decision: “…in Judaism, we say that the baby is not a life until it’s born, until part of its space is out of the body.” Multiple participants described shifts in personal priorities that shaped their decision: “…maybe it is selfish, but I didn’t want to quit my job and live in a hospital caring for a sick child.”

Most participants sought outside input in their decision making. The reported sources of input included family, friends or community, and blogs and social media. As an example of community support, one participant noted: “and his [priest’s] stance was well, just because there’s medical technology and advances doesn’t mean that you should use them on people.” Multiple families sought information about lived experiences among children with CHD, primarily through social media: “But I just knew that it [surgical intervention] wasn’t what I wanted for my child in the sense of some of the detail that people put on those blogs and journal type entries…”

Multiple participants noted a desire for resources pertaining to others’ experiences with termination or hospice birth and experiences of children living with CHD. One participant noted: “I think it would be good to have talked to another family that went through it [termination] and just to see their reasons because maybe they’re going to be different.” Overall, participants chose to pursue online content to aid with decision making, over meetings with families who chose postnatal surgery: “maybe have a site with resources… having this information right away… would make I think the decision much faster.”