Reproductive health and pregnancy experiences of women with spina bifida: A qualitative study

2.1 Methodology

A phenomenological study design was used to address this objective [23]. Phenomenology is a qualitative research approach that aims to understand others’ lived experiences through capturing their perspectives [24]. This study is part of The Pregnancy Outcomes in Women With Spina Bifida Study, an international study aimed at self-identified women with spina bifida. It was developed using the principles of person-centered research [25] in that its aims, as well as the contents of the study itself, were reviewed by individuals with lived experience through the Spina Bifida and Hydrocephalus Association of Ontario (now Hydrocephalus Canada). Several meetings occurred with individuals who lived with spina bifida in order to gain their perspective on the study and topics they wanted to see addressed. Some volunteered to provide feedback reviewed all aspects of the study including the Research Ethics Board application, the proposed methodology, and the interview guide. Their feedback was incorporated into the final study design.

2.2 Research ethics

Research Ethics Board approval was obtained through Sunnybrook Health Sciences Centre.

2.3 Research team and reflexivity

The research team consisted of five members: the principal investigator (AB; a practicing maternal-fetal medicine specialist with expertise in the field of disability and pregnancy), three senior authors (AVB: a senior medical student, SO: an academic with expertise in the field of qualitative methodology, HL: a research coordinator), and a collaborator (PC: a neonatologist and developmental pediatrician).

Reflexivity is defined as thoughtful, conscious self-awareness and is a cornerstone of qualitative research which encompasses “continual evaluation of subjective responses, intersubjective dynamics, and the research process itself” [26]. Within phenomenology, reflexivity involves evaluating the impact of one’s own experiences on the interpretation of the phenomenon under investigation [27]. Given that each member of the research team had experience working with individuals with disabilities through prior research, community partnerships, or medical practice each study team member actively sought to disentangle their subjective perceptions from their interpretation of the reproductive and pregnancy experiences of women with spina bifida through all stages of research, from pre-research to data collection to data analysis. In addition, all study team members remained aware of the existing power dynamics between researchers and participants throughout the research process. They actively requested feedback from those with lived experience, as mentioned above, in order to design an interview guide that was appropriate and sensitive to the highly personal nature of the study. In addition to reflexivity, another measure applied to enhance the study’s trustworthiness was auditability [28]. This was addressed by clearly outlining the study’s process and methodology during the pre-research stage and discussing emerging themes with the research team to ensure consensus was achieved amongst all study team members.

2.4 Recruitment

Participants were invited to participate in an internet-based, international online survey distributed by spina bifida advocacy groups and online communities including the Spina Bifida Association, Hydrocephalus Canada, and others. This anonymous online survey asked optional questions about demographics, sexual health, reproductive health, and pregnancy experiences. It was open to self-identified women over the age of 18 with spina bifida. A total of 253 people with spina bifida from 14 countries filled out the survey and 52 indicated interest in participating in an in-depth interview which was framed as an open-ended opportunity to discuss sexual health, reproductive health, and pregnancy experiences. All survey participants who expressed an interest in one-on-one interviews provided their email and were contacted with an invitation to participate. If they maintained interest, an interview was scheduled at a mutually convenient time. AVB completed the recruitment process and scheduled interviews with participants.

2.5 Data collection

A semi-structured interview guide was generated as detailed above (Table 1). AVB and HL were provided with appropriate training and conducted all interviews. Verbal consent was obtained at the beginning of every interview after describing the risks and benefits of participation. Emphasis was placed on the confidential nature of the interviews and participants were informed that there were no mandatory questions. Interviews were recorded using a TASCAM DR-05 Portable Digital Recorder and consent to record the interview was obtained from each participant before recording was initiated.

Interviews were transcribed verbatim by an external service and were anonymized by the external service who removed names of participants and any other potential identifying information. Data was analyzed by two study team members (AVB, SO) using Braun & Clark’s approach to thematic analysis [29]. They began by re-reading each transcript until they were familiar with the data set while noting initial ideas. The transcripts were imported into Dedoose software and initial codes were developed using the participants’ own language. Next, the initial codes were grouped into potential themes and the study team assessed the validity of these emerging themes. Finally, these themes were refined and presented for publication. Interviews were completed until data saturation was achieved, which was defined as the point when no novel codes or themes emerged from participant interviews [30, 31]. Data saturation was reached after the completion of 10 interviews and two additional interviews were conducted to confirm that no new codes or themes emerged.

3.1 Sample characteristics

Twelve self-identified women with spina bifida aged 28–57 (mean age 42±11 years) participated (Table 2). The majority were from North America. Within this group, four used a wheelchair, two used a walking stick, one used a leg brace, two used a combination of mobility aids (i.e. one used a leg brace and a wheelchair, and the other used a leg brace, walking stick, and a wheelchair), and three did not use any mobility aids. All 12 participants stated that their spina bifida impacted their daily function. Nine participants (75%) had experienced pregnancy. Fourteen pregnancies and 12 deliveries were described (seven (58%) vaginal, and five (42%) caesarean birth). Nine of 12 participants (75%) described their overall reproductive health experience as negative or neutral. Three of 12 participants (25%) described their overall reproductive health experience as positive.

Coding of the interviews revealed 15 themes organized into four domains: sexual health and education (three themes), pregnancy (four themes), labor and delivery (three themes), and postpartum experiences (two themes). An additional, overarching domain was social justice and advocacy (three themes). An overview of themes is available in Table 3.

Themes within the domain of social justice and advocacy were expressed by participants. These themes permeated much of the conversation surrounding reproductive health and pregnancy. They were pervasive and consistent in participants’ experiences despite participants not being asked directly about this topic. Three key themes emerged: self-advocacy, connecting and lived experience, and discriminatory attitudes.

Self-advocacy was central to participant reproductive health experiences. Many discussed advocating for their right to be pregnant and parent, seeking multiple medical opinions and searching for information. When asked about what advice they would give others living with spina bifida based on their experience, many spoke about this theme and emphasized trusting one’s own judgment, seeking information early, and asking questions.

“And it’s like, I’m like everybody else, I want to have children because I want to have children.” Participant 3 (53 years old, three pregnancies)

“. . . I think my biggest thing even with people who don’t have spina bifida is be your biggest advocate, ask lots of questions, and if you’re not getting the answers, go somewhere else to find them.” Participant 12 (49 years old, one pregnancy)

Connecting with others with similar experiences was important to participants. Some felt that people with spina bifida could provide more insight into reproductive health needs than health care providers. Online communities were a source of support for participants and many accessed resources such as Facebook to connect with other individuals in similar circumstances.

“Try to talk to a mom that has spina bifida and that has the experiences you’re looking for rather than going to a doctor.” Participant 1 (28 years old, one pregnancy)

“I couldn’t . . . find a physical community because that wasn’t in my area. But I was able to find a community on Facebook. And that was helpful just to interact with people who understood what I was going through.” Participant 5 (30 years old, one pregnancy)

. . .looking back, I wish I would have reached out to. . . people with spina bifida. Like the me today would probably seek out other people in the same situation with the same issues.” Participant 12 (49 years old, one pregnancy)

Discriminatory attitudes toward pregnancy and parenting were described by participants and were common. Some faced overt discrimination, including comments about their capacity to be pregnant and parent. Participants described people with disabilities not being seen as sexual or capable by society at large. Those who did not experience overt discrimination felt that this was not the norm.

“I feel like people with disabilities aren’t treated like sexual beings. . . and that’s negatively affected the lack of information that we have.” Participant 11 (36 years old, no pregnancies)

“. . . when I miscarried I told an acquaintance of mine. . . and she said, ^′′well, you know, that’s probably a good thing because you really can’t handle taking care of a baby, anyway. That’s probably a blessing. . . ” Participant 6 (40 years old, three pregnancies)

“I really do consider myself very lucky that I have had support, or kind of neutrality when it comes to this. I know a lot of my friends and colleagues who have a variety of different types of disabilities have faced a lot of negativity.” Participant 5 (30 years old, one pregnancy)

3.4 Sexual health and education

Within the domain of sexual health and education, three main themes emerged. The first theme included unique challenges related to sexuality, such as difficulties with bladder function, and ongoing questions about how their sexual function and spina bifida intersect.

“Since I have spina bifida, I have bladder issues as well. I remember asking a urologist about that. As far as if there was anything they could do to address bladder issues during sex, but never really got a good answer. I’ve always kind of felt like the unicorn.” Participant 11 (36 years old, no pregnancies)

“I never got specific with people about how I might be different from them in the sexual way, intimate with someone else kind of way. That’s something I still wonder about, to be honest with you.” Participant 9 (35 years old, two pregnancies)

The second revealed that sexual health was often not discussed by health care providers unless participants initiated the conversation. Health care providers who were willing to discuss sexual health were appreciated. As mentioned above, some shared how their health care providers assumed they were not sexually active.

“. . . when I think about my health care providers, as a young person, was my sexuality ever addressed, I would say definitely not.” Participant 10 (32 years old, no pregnancies)

“My doctors never really... either they never asked me much if I was sexually active. Or if they did, I kind of just felt like they were just asking a question on a sheet of paper that they had to ask kind of thing.” Participant 5 (30 years old, one pregnancy)

“I think the medical community didn’t really understand it. I think as a general societal thing, you’re disabled, nobody’s going to have sex with you. Now when I look back at it, I think that’s what was going on in people’s heads.” Participant 8 (56 years old, one pregnancy)

The third theme included the lack of sexual health resources. The majority (10 of 12, 83%) of participants did not receive sexual health information tailored to spina bifida. Many participants also described the sexual education they received in school as inadequate. Some mentioned not knowing where to seek out spina bifida-specific information, and many found support in online communities of individuals with lived experience.

“. . . it’s like they can tell you stuff they would tell everybody else, but then when it comes to. . . spina bifida it’s kind of murky waters.” Participant 11 (36 years old, no pregnancies)

“I’m not sure if I was educated on any level, except for experience.” Participant 12 (49 years old, one pregnancy)

“I’m 36, and this is the first time I’ve ever seen anyone try to even scratch the surface of this topic, let alone admit that it exists.” Participant 11 (36 years old, no pregnancies)

3.5 Pregnancy

The first theme related to pregnancy was the mixed support that participants received from health care providers. For some, a supportive health care team was the highlight of their pregnancy. Some expressed feeling lucky that they found this support. Conversely, negative attitudes were also common and incorrect assumptions were made about perceptions of the desirability of pregnancy, such as participants being encouraged to have an abortion despite this not being requested.

“Getting a team of doctors together that really knew what they were doing. . . that’s just the best thing that’s ever happened to me.” Participant 9 (35 years old, two pregnancies)

“I went to a GP, she was very gung ho on, ‘oh you’ll be wanting me to organize an abortion for you.’ Like it was very cut and dry as far as she was concerned, and I was being very selfish by considering having him.” Participant 7 (57 years old, one pregnancy)

“. . . [my doctor] was critical in helping me to feel, oh I can do this. And I should be doing this, and I have a right to be this, just like anybody else. . . I really got, I don’t want to use the word lucky. Because everyone should have the experience that I got.” Participant 5 (30 years old, one pregnancy)

The second theme related to pregnancy addressed physical functioning. Participants described several impacts that pregnancy had on their body. These included: chronic pain, incontinence and/or worsening bladder function, chronic kidney infection, swelling, and decreased mobility. Difficulty with incontinence and challenges regarding self-catheterization as pregnancy progressed were the most described. Participants were not asked specifically about tangible solutions to barriers they faced.

“Catheterizing myself became difficult. . . Not just because when you’re pregnant, you have to go to the bathroom every five seconds. But also just because my stomach was getting bigger and it was just physically harder to do.” Participant 5 (30 years old, one pregnancy)

“Because I have a Mitrofanoff. . . the most hassle part of it was as my. . . baby grew I would leak through my belly button. So I would go through five shirts a day as I got bigger.” Participant 1 (28 years old, one pregnancy)

“...[in my first pregnancy] I was hospitalized three or four times for pretty bad kidney infections. . . I basically was just on antibiotics the whole time I was pregnant the second time kind of prophylactically” Participant 9 (35 years old, two pregnancies)

The third theme related to pregnancy included a lack of information and access to health care providers experienced around disability. Participants described not being able to reliably find information about how being pregnant would impact their spina bifida, at times causing emotional distress. Many felt this was due to a lack of experienced health care providers.

“. . . the scariest thing [about pregnancy] was not having somebody that was experienced with spina bifida cases.” Participant 6 (40 years old, three pregnancies)

“I think just the emotional aspect of it. It was definitely the hardest for me. Again, just every day waking up wondering if something’s going to go wrong. . . It’s a lot harder with spina bifida. . . ” Participant 9 (35 years old, two pregnancies)

“I think [my doctor] tries to provide the best knowledge that she can, but she hasn’t had any patients with this similar situation. So it’s hard for her to provide educated knowledge and there’s not a lot of research on it in terms of outcomes. . . ” Participant 10 (32 years old, no pregnancies)

The fourth theme related to pregnancy was participants describing their pregnancy being classified as “high risk,” requiring care from maternal-fetal medicine doctors, higher appointment frequency and more screening. Participants assumed that this was due to their spina bifida. Some mentioned that their health care providers recommended invasive genetic testing due to this perceived risk.

“Because I had spina bifida, and I was admitted to a high risk [clinic]... so I had to go through all kinds of hoops that a high risk pregnancy would bring with it.” Participant 12 (49 years old, one pregnancy)

“. . . they pushed for [invasive testing], and I refused to do that, because my opinion was if my child had spina bifida, I’m just going to deal with it.” Participant 3 (53 years old, three pregnancies)

3.6 Labor and delivery

In the category of labor and delivery, the first theme was the variability of analgesia offered (Table 2). Many participants were told an epidural was not possible due to their spina bifida. Some were not told why an epidural would not be possible. Only one participant had an attempted, unsuccessful epidural. Those who had vaginal births either had no analgesia or were given intravenous medication. All those who had caesarean births were given general anesthetic. Of these participants, some described being asleep during their child’s delivery as emotionally distressing.

“I wasn’t allowed to have an epidural. They said they didn’t want to. . . make my condition worse, I guess.” Participant 1 (28 years old, one pregnancy)

“They put me out completely, they did general anesthesia, and then I had recovery time afterwards. . . My husband got to see my daughter before I did. And that was a little emotional for me.” Participant 5 (30 years old, one pregnancy)

The second theme addressed participant attitudes towards delivery methods. Some participants were eager to have a caesarean birth due to their frequent surgeries in the past. Others doubted their capacity to participate in a vaginal delivery due to decreased sensation and lower body strength.

“I was dying to have a c-section. I don’t know why but that was not as scary as natural birth because I’ve had many surgeries in the past.” Participant 9 (35 years old, two pregnancies)

“Now, the doctor I had said, “your body will do what it needs to do, and if it doesn’t we will do something different.” That was always a concern for me, just how was my body going to do it? Am I going to be able to do it?” Participant 6 (40 years old, three pregnancies)

The third theme included a sense of accomplishment that participants described after their child was delivered. For some, this was the highlight of their pregnancy experience. Participants described how people with disabilities are often seen as incapable by society at large. Therefore, proving that they were capable of giving birth felt important.

“I really think I went into it thinking, there’s no way I can do this. I can’t believe they’re having me do this, and then I did it and I felt like a superhero.” Participant 9 (35 years old, two pregnancies)

“. . . I think the best part has been being able to prove to myself and to other people that I can do this. . . the disability community is always told you can’t. But I can, and I did, and I am.” Participant 5 (30 years old, one pregnancy)

3.7 Postpartum experiences

In the domain of postpartum experiences, the first theme included the importance of emotional wellbeing in pregnancy and the postpartum period. Participants appreciated when health care providers asked them about their mental health. Some discussed the intersection between physical disability and emotional wellbeing, for example feeling guilty about giving birth due to their spina bifida.

“I felt guilty that I had him because I had my spina bifida. . . I felt like I didn’t deserve to be a mom.” Participant 4 (54 years old, one pregnancy)

“My doctors are the ones that actually recommended I see a therapist. I was really impressed with them for just even noticing that I was needing that. That’s not their wheelhouse, but they were so great and even making sure my mental health was good.” Participant 9 (35 years old, two pregnancies)

The second theme addressed unique parenting challenges that participants faced, many of which were related to mobility. Participants described coming up with creative solutions often through trial and error. Some of their infants were born prematurely and several described this being a positive outcome due to it being easier to care for a smaller child in the immediate postpartum period.

“. . . it was just concentrating on finding ways around how I was going to mother. Just the physical side of things. But I had great faith in . . . necessity being the mother of invention . . . and I was able to work it out in most cases.” Participant 7 (57 years old, one pregnancy)

“I tried wearing her when I pushed with my wheelchair. . . in a front carrier and a sling and all that. I was able to do what I needed to do with her. She was small. . . when she was born. I always tell my husband that was a blessing.” Participant 6 (40 years old, three pregnancies)