Perspectives of Young Men with Spina Bifida on Fertility Potential and Future Parenthood

Abstract

Objectives:

To explore the perspectives of young adult males with spina bifida on their fertility potential and parenthood.

Methods:

Semi-structured qualitative interviews were conducted between 2/2021-5/2021 with men ≥18 years of age with spina bifida on perspectives on fertility potential and parenthood. Demographic and clinical characteristics were obtained from a survey and chart review. Interviews were transcribed verbatim and assessed through conventional content analysis framework.

Results:

Of 30 eligible individuals approached, 20 agreed to participate. Median age was 22.5 years (range 18-29), and 80% had myelomeningocele. Most reported not being in a relationship (14/20, 70%), not being currently sexually active (13/20, 65%), and no participant reported a history of paternity. Many reported a desire for future fertility. Uncertainty and areas of concern were identified regarding their physiologic and psychosocial ability to be a parent. Perceived barriers to achieving parenthood included: 1) uncertainty around fertility potential and the hereditary nature of SB; and 2) concerns with being a parent and raising a child. Facilitators to help overcome uncertainty, such as gaining knowledge on fertility testing options and understanding means of preventing neural tube defects, were identified. Participants expressed the importance of strong peer and family support to help overcome challenges related to parenthood.

Conclusions:

Men with spina bifida are interested in future fertility, yet uncertainty exists around the physiologic and psychosocial ability to be a parent. Facilitators to help overcome uncertainty and concerns should be a focus of patient counseling around future fertility with young men with spina bifida.

Introduction

Over the last several decades, the life expectancy of individuals living with spina bifida (SB) has improved, with >80% living into adulthood.¹ Recent studies have focused on the importance of sexuality, sexual and reproductive health education, and the need to discuss such issues with individuals with SB as they age.^2,3 Interest in fertility and having biologic children has been reported in small studies among men and women with SB.^4,5 Specific to women, increases in the rates of pregnancy and successful delivery and research examining SB-specific challenges regarding pregnancy have been seen.^6,7,8 However, little remains known about fertility potential, predictors of fertility, and fertility-specific challenges faced among men with SB.

Prior studies have reported potential physiologic barriers to fertility for men with SB, yet the ability to predict fertility potential in men with SB based on phenotypic characteristics is not possible. Studies on sperm quality are limited, but small studies demonstrate impaired spermatogenesis in men with SB.⁹ Altered sexual function secondary to erectile or ejaculatory issues may also impair sperm delivery.^10,11 Potential psychosocial barriers to fertility and fatherhood include difficulty with relationship building, poor self-esteem, and anxiety around sexual functioning.^{4, 12, 13} Despite these perceived physiologic and psychosocial barriers, few studies have queried patient perspectives on their understanding of their own fertility potential and desires or concerns around parenthood. Such perspectives on fertility and parenthood, which may be conceptualized at a young age, are also not reliant on or necessarily directly linked to sexuality and sexual health, which have been explored previously in this population. There is a paucity of literature and resources for men with SB and their families to learn about their fertility and a lack of guidance for clinicians on how to counsel men with SB about their fertility. This foundational knowledge is crucial to informing better patient-centered reproductive health care and counseling and may help prioritize areas of future patient-engaged research.

Thus, we sought to perform an exploratory qualitative study to identify the perspectives of young adult men with SB around their fertility potential and interest in future parenthood. The present work was part of a larger study aimed at better understanding the perspectives of young men with SB around their sexual and reproductive health and preferences for clinician counseling and care.

Materials and Methods

Study Design and Research Team

A qualitative descriptive study was performed with young adult men with SB cared for at a large, tertiary care multidisciplinary SB center. Methodology has been described previously as this is part of a larger qualitative study.^{12, 14} Convenience sampling was employed to enroll participants in individual, semi-structured interviews. A male pediatric urology fellow who cares for men with SB and has formal education in qualitative research (JR), a female researcher with a master’s degree in public health with experience in qualitative research (IR), and a female researcher trained in qualitative research (JH) participated in data acquisition and analysis. Two pediatric urologists (DC and CS) who care for individuals with SB participated in interview guide creation. This study was approved by our Institutional Review Board (IRB 2020-3993).

Study Population

Men with SB, ≥18 years old, were recruited by telephone or at clinic visits regarding study participation. There was no upper age limit. Prior parenthood was not required for participation. Individuals aged <18 years old, non-English speaking, or deemed by their primary urologist to have cognitive impairment, limiting their ability to independently participate in interviews, were excluded. Recruitment brochures outlining study details were provided to prospective participants. A $30 gift card incentive was offered for participation.

Quantitative Measures

Demographic characteristics of participants were extracted from the medical record. A web-based, REDCap questionnaire was provided to participants to self-report their bladder management, assistive devices for ambulation, living situation, relationship status, sexual orientation, and history of sexual activity (as defined by the participant) and parenthood. Descriptive statistics were performed on demographic data.

Qualitative Interviews

Verbal informed consent was obtained, and semi-structured interviews were performed by telephone by one interviewer (JR). The interviewer had no prior relationship with any participant. Interviews were recorded and transcribed verbatim with identifiers removed. Open ended questions probed participant perspectives on fertility and parenthood (Appendix 1). Pilot testing of the interview guide was performed with a team member (JH) by the primary interviewer (JR) prior to interviews to enhance question clarity. The interview guide was adapted iteratively based on field notes taken during interviews.

Qualitative analysis

Conventional content analysis framework was used for transcript coding with MAXQDA software (VERBI Software, Berlin, Germany).^15,16 Review of three transcripts was performed in tandem for training, reliability, and development of the code book through inductive coding by two trained coders (JR and IR). A third trained coder (JH) participated in coding of the remaining transcripts. Remaining transcripts were coded either in tandem with immediate consensus or independently with subsequent re-review for consensus. Coding began by identifying words or phrases to describe and summarize the topic of discussion. Descriptive codes and phrases were placed into categories. Categories were extrapolated to broader themes. Supporting representative quotations for each theme were compiled. Participant interviews continued until no new information emerged from the study participants, suggesting thematic saturation.¹⁷ Quantification of themes was not performed, and inferences cannot be made regarding the prevalence of themes outside of the study population.¹⁸ The consolidated criteria for reporting qualitative research (COREQ) checklist was followed (Appendix 2).¹⁹

Results

Thirty individuals were approached for participation and 20 agreed to participate in semi-structured interviews (between 2/2021-5/2021). Average conversation time was 64 minutes (range 46-91 minutes). Demographic and clinical data are reported in Table 1. Median participant age was 22.5 years (range 18-29), and 80% had a diagnosis of myelomeningocele. No participant reported a history of parenthood. Most reported not being in a relationship (14/20, 70%), not being currently sexually active based on their own definition of “sexual activity” (13/20, 65%) and being heterosexual (17/20, 85%).

Table 1.

Participant demographics

Participant Characteristic	No. (%)
Median Age in Years (IQR)	22.5 (19-28)
Self-Reported Race
Asian	1 (5)
Black/African American	2 (10)
Other	5 (25)
White	12 (60)
Self-Reported Ethnicity
Hispanic or Latino	5 (25)
Not Hispanic or Latino	15 (75)
Type of Spina Bifida
Myelomeningocele	16 (80)
Lipomyelomeningocele	4 (20)
Romantically Interested in
Women	17 (85)
Men	0 (0)
Both	2 (10)
Unsure	1 (5)
Currently in a relationship?
Yes	6 (30)
No	14 (70)
Previously in a relationship?
Yes	10 (50)
No	10 (50)
History of Sexual Activity?
Yes	11 (55)
No	9 (45)
Currently Sexually Active?
Yes	7 (35)
No	13 (65)
Shunted Hydrocephalus?
Yes	15 (75)
No	5 (25)
Primary Bladder Management
CIC per urethra	15 (75)
CIC per catheterizable channel	5 (25)
Assistive Devices for Ambulationa
AFOs	7 (35)
Crutches or Braces	8 (40)
Wheelchair for distance	5 (25)
Wheelchair	5 (25)
Living Situation
Living Alone	0 (0)
Living with Parents	13 (65)
Living with other family members	2 (10)
Living with partner	3 (15)
Living with room mates	1 (5)
Living in a college dorm	1 (5)
Does participant have children?
Yes	0 (0)
No	20 (100)

^a.Several reported use of multiple devices

Important themes emerged from interviews regarding perspectives on and attitudes toward fertility, future fertility potential, and parenthood. Though several participants reported interest in learning about their fertility potential and having a strong desire to have children in the future, great uncertainty and areas of concern were identified regarding their physiologic and psychosocial ability to be a parent. Uncertainty and concern were also endorsed by those who did not have a desire to be a parent and, in many cases, drove an individual’s level of interest in parenthood. We categorized both these specific barriers to parenthood and participant-identified facilitators that helped individuals overcome areas of uncertainty and concern, with representative quotations included (Figure 1).

Figure 1.

Perceived barriers and facilitators to fertility and future parenthood with representative participant quotations.

Barriers to Achieving Parenthood

Uncertainty regarding individual fertility potential:

Several participants reported uncertainty regarding their fertility potential. One 28yo man discussed a fear that he would be unable to have children stating, “I mean it scares me ‘cause I’m almost 30 so I wanna be able to have kids now…it scares me that if I am just shooting blanks that I won’t be able to have kids.” (P5). Participants discussed their limited knowledge of fertility potential with SB and disability in general. One 26yo man reported questioning his own fertility potential, stating “people with a disability there’s kind of always sort of a question in the back of your mind…in terms of fertility…you always question it.” (P8).

Uncertainty of the hereditary nature of SB:

Many participants expressed uncertainty regarding the hereditary nature of SB and concern that they could pass SB on to their offspring. One 18yo participant had no knowledge of the hereditary nature stating, “uh I don’t know if any of my stuff is like genetically linked to like hereditary but I don’t want it to be.” (P17). Lack of education around the hereditary nature of SB was also reported, with wide reported ranges of assumed likelihood of passing SB on (Table 2). For example, one 25yo participant stated “I have no knowledge. I don’t know what…the statistics are on that, so, I would imagine they are close to fifty percent.” P22.

Table 2.

Participant perspectives on the hereditary nature of spina bifida.

Likelihood of SB being hereditary?	Representative quotations
Unsure	P8, 26yo: “It's a question I've meant to ask for probably the last five years I've seen [pediatric urologist] and I never do. [Laughing]. And I've never researched it.” P6, 28yo: “I think like there is a possibility, uh I really don't know.” P14, 23yo: ”I don't know what causes it and I don't know how likely it is if I were to have a baby, that they would also have it.” P17, 18yo: “Uh I don't know if any of my stuff is like genetically linked to like heredity but I don't want it to be.”
No more likely than the general population	P20, 26yo: “I know that…the general scientific understanding is that…spina bifida isn't genetic so…people with spina bifida that have kids…it's not an outright guarantee that their kids are going to have spina bifida or even a small chance that their kid might.” P11, 28yo: “Uh no more likely than a person without spina bifida, is what I heard, but I'm not sure if that's true.” P12, 19yo: “It can happen for anyone because it's um, what they told me is that since my parents haven't had any like medical issues or anything and they're both completely normal, that um it could happen to anyone, anyone could have a child that has spina bifida. Whether they're normal or healthy in that way or they have like a medical issue, it could happen to anyone at any time, so like it just, it's like a little surprise to you.” P15, 28yo: “If I do have a child, are they going to have spina bifida, things of that nature. Um, so the general consensus is no, I guess at this point, unless somebody's figured out something that I haven't yet.”
More likely than the general population	P16, 29yo: “Um well from what…I understand…it's more likely for them to get it because, there's a genetic component to it.” P13, 21yo: “From what I've heard, and what I've learned it would be like- like a 20 percent chance? I would think, a 25 percent chance. But like a 1 in 4 chance for uh, for a person like me to pass on my spina bifida to my children.” P22, 25yo: “I have no knowledge. I don't know what…the statistics are on that, so, I would imagine they are close to fifty percent.” P19, 28yo: “And, well, basically from there on I know, there's a 50% chance, I mean if I have like a child of my own, it could have spina bifida itself.” P21, 29yo: “Um, my understanding of it, it is a high possibility.” P10, 20yo: “I'm almost for certain that- that would be the case… you know I have my genes, I have my genetics, so there's really- it's really a high chance that that will happen.”

Concerns with having a child with SB:

Due to the personal struggles faced by participants, concern regarding having a child who also has SB was reported. While not a sentiment shared by all, one 28yo participant was conflicted about having a child who could also have SB, stating “I’ve always worried about bringing a life into the world that is gonna have to suffer like I did…I’m uncomfortable with creating a person with spina bifida.” (P5). Participants expressed concerns with being a parent to a child with a disability, with one 26yo participant stating, “I know parents that…are pretty much caretakers…that’s a tough thing to do in your sixties…that’s just, a tough burden.” (P8).

Concerns with being a parent with a disability:

Several participants expressed general concerns with being a parent regardless of whether their child had SB. These concerns were specific to living with a disability, with one 20yo participant stating “I don’t necessarily want kids of my own. And the reason why is because I feel like me and my disability and…what I have to take care of…myself…I feel like doing that along with taking care of another person…would be too much on me.” (P10). Others expressed concerns with raising a child, with one 28yo man reporting that “there’s also the normal, um, angst of raising a kid…will I be able to feed them, will I be able to hold them, all those sorts of fears.” (P5).

Facilitators to Overcome Concerns and Uncertainty

Despite the several themes around uncertainty and concern, participants reported optimism and highlighted several areas that could help people living with SB overcome the uncertainty and concerns that they may have around fertility and parenthood.

Tests and procedures to assess and achieve fertility:

Participants acknowledged that there are ways to determine fertility potential. One 20yo man expressed uncertainty regarding his own fertility potential but stated “I guess the only way to know would be testing.” (P3). Despite the uncertainty regarding the ability to ejaculate and produce viable sperm, one 26yo participant expressed optimism, highlighting that “there’s always a procedure for me to be capable of reproducing.” (P21).

Education around SB prevention:

Many participants acknowledged that they had learned about ways to decrease the risk of neural tube defects, notably folic acid supplementation. One 26yo participant emphasized that those wishing to carry a pregnancy should “take folic acid, whatever you do, take folic acid. Because the chances go down…from what I understand, pretty significantly if prenatal-wise, you take folic acid.” (P8).

Personal experiences of living with SB:

Though there were concerns about having a child who also had SB, several participants discussed how their experiences of living with SB would make it easier if their child also had SB. One 28yo participant felt that he could act as a support system stating, “I’d probably be one of the ones that somebody with spina bifida would want to talk to, to you know, kind of work through some things and shed light on some of the questions that they may have living with it.” (P15). Another 19yo participant felt that he could teach his child about how to best live with SB, including how to access activities like adaptive sports. He stated that “if my child were to have spina bifida, [I’d] like teach them like the things I went through and what to do or what not to do.” (P12).

Peer and family support:

Participants stressed the importance of having strong family and peer support to help overcome the challenges of raising a child. One participant said, “I have a great family, so if something like that were to happen to me, I have support.” (P11). Another 26yo participant reported that “over the years, I’ve met older people with spina bifida who, you know, have had healthy relationships and have kids” (P20). This served as a positive example for his potential future with a partner and children of his own.

Comment

In this exploratory qualitative study, we identified perspectives of men with SB around future fertility and parenthood, highlighting areas of uncertainty and concern but also elements of optimism and preparation. Our findings demonstrate that many men with SB have limited and varying degrees of knowledge about their fertility potential. Providing education on fertility potential, ways to test for fertility, and the hereditary risk of SB is likely needed in the clinical setting, as young men with SB may not be receiving such information elsewhere. While not all participants expressed desire to be a parent, several did. Assisting in connecting young men with SB to peer support opportunities may allow for increased optimism about the ability to be a parent in the future.

Prior studies have demonstrated that while many with SB receive sexual and reproductive health education, it is often deemed inadequate.^4,20 Participants in our study expressed limited formal education on fertility in school, with no focus on fertility as it relates to SB. Sexual and reproductive health, while closely intertwined, have unique aspects to them, and it is possible that a greater focus is placed on education around sexual health and function in this population.²¹ Discussions about reproductive health with individuals with SB are also rare in health care settings.³ Among young adults with SB, men have reported less commonly receiving education on fertility compared to women.¹³ Participants in our study reported anxiety regarding their own fertility potential and general lack of knowledge. Though challenging, clinicians can reassure individuals that fertility testing is possible and that there are multiple ways to achieve parenthood.

While many participants reported a desire to have biologic children, this was not unanimous. The fear of the hereditary nature of SB and worry of having a child who also has SB was often a driving force for those with no desire for parenthood. Men expressed varying degrees of uncertainty around the hereditary nature of SB and provided a range of estimates of the likelihood of passing SB on to offspring. This finding is consistent with one prior study where roughly half of participants with SB or cystic fibrosis believed there would be an increased chance of their child also having their condition.²² The exact likelihood of the offspring of males with SB also having SB is not known, possibly due to the low paternity rate in this population. Prior literature suggests an increased risk of neural tube defects in the offspring of individuals with SB similar to that of a second child of a family who have already had one child with a neural tube defect (~4% risk).²³ Though potentially causative individual genes have been identified, evidence suggests that SB and other neural tube defects occur secondary to several genetic, epigenetic, and environmental factors.²⁴

There is a clear link between maternal nutritional status and the risk of neural tube defects, with folate deficiency being the most strongly supported association.²⁵ For those with prior pregnancies with neural tube defects or for women with SB, 4 mg/day of folic acid supplementation is recommended when pregnancy is intended.^{16, 26, 27} Those participants who discussed knowledge of folic acid supplementation as a means of reducing neural tube defect incidence viewed this knowledge as reassuring and important to review with their clinicians. Few participants reported discussing the hereditary risk of SB with their clinician, which is consistent with prior literature, and may be an opportunity for improved clinician communication.²²

Participants in our study not only expressed concern with having a child with SB from the perspective of burden to the child, but also expressed concerns about the ability to parent. Caregiver/parental burden, burnout, and expressed emotion have been reported in prior studies of caregivers of youth with SB and other special needs.^28,29 Specific challenges of caring for an individual with SB include activities of daily living, time consumption, financial burden, and strain on social and familial relationships.³⁰ Identifying social support for caregivers has been shown to improve stress and mental satisfaction.³⁰ Participants in our study expressed that having family support would make raising a child with SB easier. Several participants expressed that their own experience of living with SB would make it easier for their future children if they were to also have SB.

Challenges related to being a parent while living with a physical or intellectual disability has been explored, with several unmet parental needs regarding child rearing, meeting personal needs while parenting, and navigating the health care system identified.³¹ Occupational therapists may be able to provide valuable parental assessments to identify resource needs (e.g., additional adaptive equipment) to ensure safe childcare can be provided.³² Further work is needed in this field to best understand the support that individuals with SB require to successfully perform self-care and serve the role of a parent.

Through this study, we have identified several recommendations for clinicians caring for young adult males with SB. Discussing folic acid supplementation, the hereditary risk of SB, fertility potential, and ways to assess fertility are important. Clinicians have an opportunity to ease anxiety, provide additional educational resources, and offer referrals to other clinicians with expertise in male fertility. Clinicians should routinely discuss safe sex practices with men with SB, which includes education on contraception and the use of latex free condoms given high rates of latex sensitivity among individuals with SB. Discussing safe sex practices may naturally lead to discussions of fertility and pregnancy. Specific to parenthood, identifying peer support opportunities with individuals with SB who have become parents and leveraging care partners such as occupational therapists for needs assessments may be helpful for those who desire to be a parent.

Our study has strengths and adds to the paucity of literature on male fertility in SB. The use of individual interviews allowed for in-depth descriptions of sensitive lived experiences, which would have unlikely been obtained through other methodologic approaches. Our study has limitations. Participants were recruited from a single, large, Midwestern, tertiary referral center and our findings may not be transferable to other populations. Differences regarding prior sexual experience, relationship history, sexual orientation, and gender identify may differ between our study participants and the broader population of males with SB. We excluded individuals whose primary language was not English, so may have missed cultural influences on reproductive health. Our study was limited to men over age 18, and not intended to reflect the experiences and preferences of adolescents. Cognitive and intellectual ability was not formally assessed prior to performing interviews. Thus, conversations may have been limited by incomplete question understanding by participants. No participant was a parent, thus perspectives of those who have achieved parenthood are lacking. Last, there is potential for reflexivity bias in the interviewers and self-selection bias, response bias, or non-response bias among participants. The potential limitations of this study are mitigated by the diverse population in terms of types of SB, disability severity, living arrangements, prior sexual experiences, and sexualities.

Potential next steps to this work include development of more comprehensive patient and family education materials on fertility potential, hereditary risk of SB, and resources for fertility testing. Ultimately, a large, population study assessing semen parameters will be needed to correlate phenotypic features with spermatogenesis. This knowledge would greatly assist counseling on future fertility potential for patients and families of children born with SB.

Conclusion

Young men with SB are interested in learning about their fertility potential and in some cases wish to be a parent. However, uncertainty exists around the physiologic and psychosocial ability to be a parent. Clinicians can support individuals with SB during fertility related discussions by focusing on patient perceived facilitators to overcome uncertainty and concern around future fertility and parenthood.

Appendix 1: Interview Guide

Sex/Fertility Education:

Can you tell me about when you first learned about sex?

Was it the right time to learn about sex (too late, too soon)?

How did you learn about it?

Probes:

• From parents, doctor, siblings, peers, school sex ed, internet, never learned?

• What was good about the way you were taught or learned?

• What do you think could be changed/made better?

• Are there other ways you wish you would have learned about sex?

Can you tell me whether you felt that what you were taught applied to you?

Probes:

• How directed to people with spina bifida were the things you learned?

• How could it have been better for you?

• Can you tell me about any conversations you’ve had about how sex may or may not be different for you having spina bifida?

What have you been taught about having sex with a physical disability?

Probes:

If taught:

• Can you tell me more about what you were taught?

• What modifications were discussed?

• How well prepared for sex do you feel?

• What else would you like to know?

If no:

• How well prepared for sex do you feel?

• What things do you want to know about having sex with a physical disability?

What things have you learned about having babies?

Probes:

• Are the other things you wish you would have learned?

• How well did this apply to people with spina bifida?

• Can you tell me about what you’ve been told about how having babies may or may not be different for you with spina bifida?

Discussions with your doctor:

Tell me about any conversations you’ve had with doctors about sex?

Probes:

• How did this conversation go?

• Was it comfortable or uncomfortable?

• What made the experience comfortable/uncomfortable?

• How would you describe the experience?

• What do you think was good about it? For example, were the things you learned particularly applicable to you?

• What things do you think could have been better?

• Who started the conversation?

• If brought up by provider: How difficult would it be for you to bring up the topic?

• Who was there for the conversation? (alone vs with parents)

• What type of doctor did you talk to (PCP/pediatrician, urologist, etc)?

• How old were you when this occurred? Was that the correct time for you to have that discussion?

• Is there anything you wish you could talk about but haven’t?

• Was there anything that prevented you from having these conversations? (parents present, uncomfortable environment, other more important medical issues?)

How interested in being sexually active were you when you had these conversations?

Had you already been sexually active prior to these conversations?

IF NO PRIOR CONVERSATIONS:

How do you feel about the fact that you haven’t talked to your doctor about this?

Probes:

• Why do you think this hasn’t been discussed?

• What things would you want to ask or know more about?

• Who should start these conversations?

• Who should be in the room during these conversations?

• At what age should these conversations be had?

Tell me about any conversations you’ve had with doctors about having children?

Probes:

• How did this conversation go?

• Was it comfortable or uncomfortable? What made the experience comfortable/uncomfortable?

• How would you describe the experience?

• What do you think was good about it? For example, were the things you learned particularly applicable to you?

• What things do you think could have been better?

• Who started the conversation? If brought up by provider: How difficult would it be for you to bring up the topic?

• Who was there for the conversation? (alone vs with parents)

• What type of doctor did you talk to (PCP/pediatrician, urologist, etc)?

• How old were you when this occurred? Was that the correct time for you to have that discussion?

• Were you interested in being sexually active then or had you already been sexually active?

• Is there anything you wish you could talk about but haven’t?

• Was there anything that prevented you from having these conversations? (parents present, uncomfortable environment, other more important medical issues?)

IF NO PRIOR CONVERSATIONS:

How do you feel about the fact that you haven’t talk to your doctor about this?

Probes:

• Why do you think this hasn’t been discussed?

• What things would you want to ask or know more about?

• Who should start these conversations?

• Who should be in the room during these conversations?

• At what age should these conversations be had?

What have you been told about your ability to have children of your own in the future? (from doctor or others)

Probes:

• How did these conversations go?

• What was good about it?

• How could it have been better?

Are there things you wish doctors had or would discuss with you about sex?

Are there things you wish doctors had or would discuss with you about having children?

What suggestions do you have for doctors when talking to patients about sex?

Probes:

• Who should bring up the topic?

• What should they talk about?

• When should they bring it up?

• Should parents be in the room or out of the room?

• How can they make it comfortable to discuss these topics?

What suggestions do you have for doctors when talking to patients about having children?

Probes:

• Who should bring up the topic?

• What should they talk about?

• How can they make it comfortable to discuss these topics?

Seeking counseling:

If you wanted to have a baby, who would you talk to about it?

Probes:

• From parents, friend, partner, doctor

• Would you talk to any of your doctors about the decision?

• Where would you go for more information?

What is your experience with friends or acquaintances with SB who have had children?

Probes:

• If you know someone, did they seek help from doctors?

What concerns do you have about getting a partner pregnant or having your own children?

Probes:

• For example, the physical act of getting a partner pregnant vs caring for children given physical disability

• How do you feel about finding a partner?

• What is your understanding of the likelihood of your baby having spina bifida? What does that mean to you?

Interest in sexual activity/future fertility:

What does being sexually active mean to you?

What opportunities have you had to be sexually active?

Probes:

If been active:

• What sort of sexual activity did you have (kissing, touching, oral sex, intercourse)

• What was good about it?

• What was bad about it?

• What sort of conversations did you have with your partner before?

What advice would you give others with spina bifida about being sexually active?

• How interested are you in being sexually active in the future?

If never been active:

• How interested are you in being sexually active?

• If not interested: How do you think that may change with time?

• Is there anything that may change this for you? (ie having a partner)

• If interested: What do you think has prevented you from being sexually active?

• How do you think that may change with times?

• Is there anything that may change this? (ie having a partner)

• Are there particular things that worry you about this? Physical, psychologic, issues with relationships, concern about ED

**For those who have an have not been sexually active in the past**

Define Intercourse for the participant: sexual intercourse refers to penetrative sex, or entering a partner

How confident are you in your ability to have sexual intercourse if you wanted?

Probes:

• What are you confident about?

• What things are you unsure of? (ie mobility, finding a partner, living situation, urinary leakage, functional difficulties)

• What worries do you have about this?

How do you think having sex or having a relationship may be different for you compared to someone without SB?

Probes:

• What about sex may be different?

• Is there anything that you think would prevent you from having sex or a relationship?

How happy are you with your erections?

Probes:

• If unhappy: Have you considered medications? Has anyone ever told you that medications may help? Has this prevented you from being sexually active?

• What sort of help have you asked for if any?

• How confident are you in your ability to have an erection?

Ejaculation refers to the ability to eject semen from your penis. Are you able to ejaculate?

Probes:

If yes:

• How happy are you with your ejaculations?

• What is that experience like for you?

• How confident are you in your ability to ejaculate?

If no:

• How does that impact you?

• How does that impact your sexual experiences?

• How does that impact your partner?

An orgasm is the feeling of sexual pleasure or reaching a climax. Are you able to orgasm?

Probes:

If yes:

• How happy are you with your orgasms?

• What is that experience like for you?

• How confident are you in your ability to orgasm?

If no:

• How does that impact you?

• How does that impact your sexual experiences?

• How does that impact your partner?

What did you think about the questionnaire you completed asking about your erections, ejaculation, etc. (IIEF)?

Probes:

• How well did you understand the questions?

• How well did it apply to you?

• What do you think was left out regarding how you experience sex?

• What other things do you think should be asked?

• What could have made it better?

Have you thought about having children of your own, even if you don’t want them now? Can you tell me your thoughts about this?

Probes:

If not Interested:

• Why do you think that is?

• No interest, sexual dysfunction, barriers to relationships?

If interested:

• Have you tried to have children of your own?

• How has that gone?

• If children: What was your experience like? Did you have any difficulties getting pregnant?

• What worries do you have about this?

• How would you feel about discussing this with your doctor?

Closing Statement:

Is there anything else that you would like to share that we haven’t discussed?

Is there anything you would like to tell your doctors or wish they knew?

Is there anything else you want us to know about you?

Appendix 2: COREQ-32 Checklist

Topic	Item No.	Guide questions/description	Reported on Page #
Domain 1: Research team and reflexivity
Personal Characteristics
Interviewer/facilitator	1	Which author/s conducted the interview or focus group?	3
Credentials	2	What were the researcher’s credentials? E.g. PhD, MD	Primary interviewer (JR) is a medical doctor (MD)
Occupation	3	What was their occupation at the time of the study?	2
Gender	4	Was the researcher male or female?	2
Experience and training	5	What experience or training did the researcher have?	2
Relationship with participants
Relationship established	6	Was a relationship established prior to study commencement?	3
Participant knowledge of the interviewer	7	What did the participants know about the researcher? e.g. personal goals, reasons for doing the research	3, included in recruitment brochure
Interviewer characteristics	8	What characteristics were reported about the interviewer/facilitator? e.g. Bias, assumptions, reasons and interests in the research topic	3, included in recruitment brochure
Domain 2: Study Design
Theoretical framework
Methodological orientation and Theory	9	What methodological orientation was stated to underpin the study? e.g. grounded theory, discourse analysis, ethnography, phenomenology, content analysis	3
Participant selection
Sampling	10	How were participants selected? e.g. purposive, convenience, consecutive, snowball	2
Method of approach	11	How were participants approached? e.g. face-to-face, telephone, mail, email	2
Sample size	12	How many participants were in the study?	4
Non-participation	13	How many people refused to participate or dropped out? Reasons?	4
Setting
Setting of data collection	14	Where was the data collected? e.g. home, clinic, workplace	Interviews were performed in a private office
Presence of non-participants	15	Was anyone else present besides the participants and researchers?	3, Participants were asked to be alone, in a quiet, private location
Description of sample	16	What are the important characteristics of the sample? e.g. demographic data, date	4
Data collection
Interview guide	17	Were questions, prompts, guides provided by the authors? Was it pilot tested?	3
Repeat interviews	18	Were repeat interviews carried out? If yes, how many?	No
Audio/visual recording	19	Did the research use audio or visual recording to collect the data?	3
Field notes	20	Were field notes made during and/or after the interview or focus group?	3
Duration	21	What was the duration of the inter views or focus group?	4
Data saturation	22	Was data saturation discussed?	4
Transcripts returned	23	Were transcripts returned to participants for comment and/or correction?	Transcripts were not provided to participants
Domain 3: Analysis and Findings
Data analysis
Number of data coders	24	How many data coders coded the data?	4
Description of the coding tree	25	Did authors provide a description of the coding tree?	4
Derivation of themes	26	Were themes identified in advance or derived from the data?	4
Software	27	What software, if applicable, was used to manage the data?	3
Participant checking	28	Did participants provide feedback on the findings?	Feedback on final themes was not provided by participants
Reporting
Quotations presented	29	Were participant quotations presented to illustrate the themes/findings? Was each quotation identified? e.g. participant number	Figure 1, Table 2
Data and findings consistent	30	Was there consistency between the data presented and the findings?	5-8
Clarity of major themes	31	Were major themes clearly presented in the findings?	5-8
Clarity of minor themes	32	Is there a description of diverse cases or discussion of minor themes?	5-8