Understanding health-related quality of life after hypospadias repair: A qualitative study with pre-adolescent males and parents

Summary

Introduction

Hypospadias is a common disease that affects approximately 1 in every 200 live male births in the United States, and long-term studies of individuals who have undergone repair demonstrate complication rates of 15%–70%. The Hypospadias-Specific Health-related Quality of Life (HRQOL) Conceptual Framework for youth and adults suggests that additional morbidity may be incurred from poor psychological, social, and sexual health. The current study sought to clarify hypospadias-specific HRQOL and care priorities in a pre-pubertal population.

Material and methods

This IRB-approved, semi-structured interview study used rigorous qualitative research methods. Eligible patients were English-speaking 8–12-year-old males with hypospadias and their parents. Families completed a demographic questionnaire and separate youth and parent 30-min telephone interviews. We used hybrid thematic analysis to develop an operational codebook, analyze participant responses, and generate conceptual themes. Mixed methods analysis was used to explore patterns of experiences across groups defined by socioeconomic level.

Results

We interviewed 10 parents and 8 children (Median age 9 years, Range 8–11). We generated three overarching themes: Penile Factors, Psychosocial Concerns, and Expectations of Surgery and the Healthcare Team. These highest-order themes were generated for youth, parent-proxy, and parent self-reported experiences, and there were different sub-themes for each participant type (Figure). Youth were focused on avoidance of disclosure and the psychological impact of self-comparisons and embarrassment, while the parental perspective centered on worries about future fertility, complications, psychological health, and normality. Some youth and parents from disadvantaged neighborhoods or those with public insurance indicated a need for more education on normal penile functions and provision of strategies for long-term self-monitoring and facilitation of long-term follow-up on mixed methods analysis.

Conclusion

These findings add insight into the multifaceted experiences of pre-pubertal youth and families dealing with hypospadias, and underscore the consistent, wide-ranging interplay between medical, psychological, and social concerns. Patterns in themes across socioeconomic status and insurance coverage suggest that access to information and quality care may vary significantly and could contribute to health disparities. Urologists should employ an individualized approach to counseling and care delivery. Future studies will seek to characterize care priorities in pubertal and post-pubertal age groups to design developmentally adjusted support tools for youth and adults with hypospadias and their families.

Introduction

Hypospadias affects approximately 1 in every 200 live male births in the United States [1]. Long-term studies of patients who have undergone distal or proximal repair demonstrate complication rates of 15–70% [2–5]. Emerging studies using patient-reported outcome measures (PROMs) suggest that additional morbidity is incurred from poor psychosocial or psychosexual health [2–4,6–14].

We previously developed the Hypospadias-Specific Health-Related Quality of Life (HS HRQOL) Conceptual Framework based upon thematic coding of literature from PROM assessments of youth and adults with hypospadias. This framework utilized the World Health Organization’s holistic definition of HRQOL and identified five specific domains, including penile appearance, voiding function, social function, psychological health, and sexual health [11,15]. Less research has used qualitative methods to describe the lived experiences of people with hypospadias and their families. To develop age-adjusted support tools for youth and adults with hypospadias, our goal was to explore our framework’s applicability to patients in different stages of development through qualitative evaluation.

The aim of this qualitative study of pre-pubertal youth and families of youth with hypospadias was to identify cross-cutting and role-specific themes to support or refine the Hypospadias Specific HRQOL Conceptual Framework and guide HRQOL-informed care for youth with hypospadias.

Methods

This IRB-approved study conducted at a tertiary care pediatric hospital in the Southwestern United States enrolled English-speaking 8–12-year-old males with surgically-corrected hypospadias and their parents; those with medical or neurocognitive comorbidities that precluded autonomous patient interviews were excluded. Study staff called families from a list of potential eligible candidates who had been identified using ICD-10 and CPT codes, to explain the study and obtain parent consent and child assent. For this study, our goal was 20 families, or sufficient interviews to achieve thematic saturation.

Measures

Parents completed a demographic questionnaire, and investigators collected data on hypospadias severity, surgical correction, and complication rates. Using participant addresses, we calculated the area deprivation index (ADI), a valid measure of socioeconomic disadvantage based on census data. The ADI defines a state decile of >5 and a national centile of >50 as a socioeconomically disadvantaged neighborhood [16].

Youth and parents completed separate 30-min telephone interviews with one investigator (VPB). The interview introduction established that youth and parents were interviewed as the experts on life experiences and care priorities related to hypospadias. The semi-structured, open-ended interview was designed based upon HS HRQOL Conceptual Framework [11]. Our format elicited youth and parent experiences related to various aspects of living with hypospadias, as well as prompts to clarify questions and probes to encourage additional discussion. See Supplement for interview scripts.

Data preparation

Interviews were deidentified, audio-recorded and transcribed verbatim with a HIPAA-compliant professional medical transcription service (Landmark Associates, Inc; thelai.com). The research team reviewed transcripts for accuracy prior to analysis.

Data analysis

We used hybrid thematic analysis to identify common concepts that described patient and parent experiences in hypospadias. Two authors (VPB, MEH) authors read 3 transcripts to develop a codebook with standard definitions. One author applied the codebook to the transcripts and iteratively revised the codes and definitions as new concepts were observed. All transcripts were reviewed using the final codebook [17]. Interviews stopped once no new information was obtained. The second author (SC) separately coded 5 interviews using the final codebook, and discrepancies were reviewed and resolved by the senior author. Two authors (VP, AP) conducted second-round coding to consider code relationships [17].

The full investigator team, comprising expertise in qualitative research, pediatric urology, and pediatric psychology, reviewed codes and potential associations to generate themes. Themes were considered in relation to our previously developed conceptual framework. We used NVivo 12 software for all qualitative analyses [QSR International, 2020).

Mixed methods analyses were performed to compare response patterns based upon age at initial surgery, complication rates, social determinants of health, including ADI and insurance status [16]. Fisher’s exact analyses were used to compare the types of hypospadias and complication rates within different demographic groups, with a p < 0.05 designated as statistically significant.

Results

Participant characteristics

We attempted to contact 65 potentially eligible families, of which 19 were successfully contacted for recruitment. Of those, seven did not meet eligibility criteria, 1 parent did not feel that hypospadias impacted their child’s quality of life, and 1 was uncomfortable with their child being interviewed. The remaining ten (52.6%) consented to participate and completed interviews before thematic saturation was reached (10 parents, 8 children). Six families (60%) came from disadvantaged neighborhoods, and 5 (50%) had public insurance. Demographics and participant surgical details are available in Table 1 and Table 2. Of note, the distribution of hypospadias severity (p = 0.33) and rate of treatment for complications (p = 0.52) was not significantly different between families from disadvantaged or advantaged neighborhoods. Similarly, hypospadias severity (p = 0.99) and rate of treatment for complications (p = 0.99) was not different between families with public or private insurance. There was no difference in hypospadias severity (p = 0.444 for youth who did or did not have complications requiring reoperation.

Table 1.

Demographics of study population.

Demographics	No. Patients (%)
Age at Initial Hypospadias Repair (Median yrs, IQR)	2 yrs (6)
Age at Interview (Median yrs, IQR)	9 yrs (2)
Race
Black or African American	5 (50%)
White	3 (30%)
Asian	1 (10%)
Other	1 (10%)
Ethnicity
Hispanic/Latino	2 (20%)
Non-Hispanic/Latino	8 (80%)
Health Insurance
Public Insurance (Medicare/Medicaid)	4 (40%)
Private	5 (50%)
Not Reported	1 (10%)
Area Deprivation Index
≤ 5 (less disadvantaged)	4 (40%)
> 5 (more disadvantaged)	6 (60%)
Hypospadias Location
Glanular	3 (30%)
Distal	3 (30%)
Midshaft	2 (20%)
Penoscrotal	1 (10%)
Perineal	1 (10%)
Hypospadias Class
Distal	8 (80%)
Proximal	2 (20%)
Complications (fistula, glans dehiscence, meatal stenosis, diverticulum)
Yes	5 (50%)
No	5 (50%)

Table 2. Surgical Details By Patient.

Participant ID corresponds to Parent or Participant Quotes in Table 3.

Participant ID	Age at Surgery (years)	Age at Interview (years)	Location of Hypospadias	Number of Planned Surgeries	Number of Complications	Number of Total Surgeries
A	2	9	Glandular	1	0	1
B	9	10	Midshaft	1	1	2
C	1	10	Perineal	2	1	3
D	2	9	Glandular	1	1	2
E	5	11	Distal Shaft	1	1	2
F	0.75	8	Distal Shaft	1	0	1
G	10	10	Midshaft	1	0	1
H	1	8	Penoscrotal	1	4	5
I	7	8	Glandular	1	0	1
J	0.5	8	Distal Shaft	1	0	1

Qualitative themes

The research team generated three overarching themes: Penile Factors, Psychosocial Concerns, and Expectations of Surgery and the Healthcare Team. These highest-order themes were common across the three roles: youth discussing their own experiences, parents discussing youth experiences (“parent-proxy”), and parents discussing their own experiences. Each theme had unique sub-themes for the 3 roles, as illustrated in Fig. 1, with example quotes in Table 3.

Fig. 1. Thematic Framework for Health-Related Quality of Life (HRQoL) in Youth Ages 8–12 and Parents of Youth with Hypospadias.

Codes are denoted as follows: Y-Youth, PP-Parent-Proxies, P-Parents. Three main themes include Penile Factors, Psychosocial Concerns, and Experiences with Surgery and the Healthcare Team. Unique codes are listed under each main theme. Overlapping codes between two main themes are indicated with a single line extending from the area of overlap. The codes overlapping between all three main themes are indicated by the star in the center of the figure.

Table 3. Illustrative Quotes for Subthemes.

Sample quotes for each subtheme are provided in the table below. Quotes from parents and youth in the same family are indicated by the same letter (i.e. Participant A corresponds to Parent A).

Theme 1: Penile Factors
Sub-themes	Sample Quote
Youth Experiences
Penile Function	When I go pee sometimes, um, it comes out in like little chunks, and I try and try. And when it doesn’t feel like I need to go anymore, I wash my hands and leave the bathroom and all. And I start feeling like it again, and it does the same thing. (Participant A)
Appearance
• Satisfaction	… [To] me, I think it looks fine. (Participant B)
• Discomfort	I feel pretty uncomfortable [about how my penis looks right now]. It’s hard to explain … but I have a little hole in it – it doesn’t ….squirt straight. (Participant C)
Urinary Spraying	When I’m trying to … go to the restroom, it’s hard to hold. (Participant D) Sometimes pee gets on my feet when I’m in the shower. (Participant A)
Pubertal Development	I started growing—feeling really painful. But then next day was a little bigger, and I couldn’t sleep through the night so I went to wake up my mom about it, and she says it’s this process where, um, I’m just starting to grow and get older. (Participant A)
Parent-Proxy Descriptions of Youth Experiences
Penile Function
• Erections	It is happening, and he wondered why. And … I didn’t have a medical answer for him. (Parent B)
• Urinary Control Problems	… the only – problem,…that he had was not being able to pee, you, know, without side stream … so … going to the bathroom was messy. (Parent E)
Penile Appearance
• Satisfaction	… It doesn’t bother him in any sort of way. Like, the look and everything seems to be fine. … (Parent F)
• Dissatisfaction	… [H]e always expressed that he did feel that his, uh, you know, his penis looked weird …. (Parent G)
• Self-Comparisons	He has, in the past, asked me, “Mom, why do I look like this? And why is it—why do I not look like <Name>?.. I wanna look like that.”” (Parent H)
Embarrassment	Since he’s older, um, he- he feels somewhat embarrassed, only regarding the size. (Parent C)
Parent Experiences
Penile Size
• Comparison	… as of today, I still feel as if, um, it’s small for his age. (Parent C)
• Importance	[I only ever worry] … growth-wise. You know, ’cause you know you want him to have a normal healthy, you know, life as far that–that aspect. (Parent I)
Urinary Control
• Importance	I don’t want him dealing with incontinence as he gets into, you know, like, middle school and high school. (Parent H)
• Toilet Hygiene	He might try to aim, and … you know, have an accident, but he pretty much knows, um, the- the process in regards to keeping the toilet clean.(Parent C)
Theme 2: Psychosocial Function
Subcodes	Sample Quote
Youth Experiences
Social Functions
• Public Exposure	I feel fine [using the bathroom at school, as long as nobody is watching. (Participant A)
• Thoughts About Disclosure	… I’ve told them I’ve had surgery … only my friends, so I would say that’s about 11 or 10 people. (Participant G) [I told] only my friend that I’ve known my whole life. (Participant H) [I talk to] only my mother [about hypospadias]. (Participant B)
Attractions	[Hypospadias] does not affect anything [about crushes]. (Participant G)
Future Worries	[I worry] that maybe that, um, that [hypospadias will] just stay forever, maybe. (Participant I)
Trauma from Surgery	Whenever it is—once I’ve had with these five surgeries in my lifetime— it makes me feel like when I didn’t look that something could happen to me. (Participant H)
Complications	… [T]here was one time where they said I might have to have surgery done again … They said it may [be] a painful process, of course … It started making me worry about it. (Participant A)
Parent-Proxy Descriptions of Youth Experiences
Interpersonal Interactions
• Disclosures to Friends	Um, I think he has actually shared it with only a couple of his really close friends, but—uh, they’ve mostly been supportive. (Parent G)
• Romantic Interest/Relationships	Yes … he’s had … a girlfriend … and things like that. So he’s definitely had some interest in it. (Parent A) Uh, he has never expressed any interest in any male or female period … in his own peer group. (Parent G)
Public Restroom Concerns	He just, uh, that’s just a fear he’s always had, [that] something’s wrong … he feels like the whole surgery wasn’t … I think in a lotta ways he felt embarrassed about, uh, going to the bathroom at school. (Parent G)
Trauma from Surgery	Pulling [the catheter] out was rather painful. And for the next few days, he was in a lot of pain. I mean, he was—It was really very sad. He, um, you know, cried and said, “Why did you do this to me, Mommy?”. (Parent E) They pulled his Foley out, …[and] I had to wait until he voided—before leaving. And my son is screaming in excruciating pain … for about 2 h, he suffered. So, that was, uh, very traumatic. (ParentJ)
Social Disadvantage	[I don’t know if this is because he was in] foster care, um, and social services, but he, um, he just, uh, that’s just a fear he’s always had, [that] something’s wrong … his doctor was very well aware that he had this condition and always said it didn’t need to be fixed. (Parent G)
Parent Experiences
Growth and Development
• Peer comparisons	So far, I think he’s on par with everyone else. (Parent A) He’s very—um, he’s always from six months to maybe even two years physically less mature than everyone else. (Parent E)
• Sex and Gender	Um, they thought, well, maybe he was developing into a girl [in utero]. You know? And then certainly became a boy … But maybe he’s, you know, genetically a female, not necessarily a boy. You know? (Parent J)
Stigma and Disclosure	Like I said, [his surgery is] something that’s private with just between me and him so … (Parent I) So don’t nobody really know–know of his surgery … (Parent J)
Thoughts About Public Exposure	… most kids … did the stand-up urinal. And I would tell ‘em, “No. Go to the closed one.” (Parent F)
Sources of Worry
• Fertility/Function	So, one of my concerns for him, in the future, if he decides to get married and have children—or let me just say, if he decides to father children, [laughter] that it’ll be hard for him. (Parent B)
• Complications	[I worry about …] if he needed an additional surgery, because, you know, things get bigger, things move, hormones—whatever. That’s what I’m kinda concerned about. (Parent A)
• Embarrassment	We just want it to be normal size and functioning, so he doesn’t feel embarrassed. (Parent C)
• Normality	I was concerned about, like, maybe the bottom part being a little saggy. And she’s like, “That is nothing to concern yourself about. That looks normal.” I’m like, “Okay.” (Parent F) I think it looks normal, just not, um—I’m just concerned with the—I don’t know how it should look as far as the growth. (Parent I)
Theme 3: Expectations of Surgery and the Healthcare Team
Sub-themes	Sample Quote
Youth Experiences
No Knowledge of Surgery	… even I didn’t even know I had it. (Participant F)
Benefits of Surgery	… they put the hole where it was supposed to be, it helped me. (Participant G)
Uncertainty	I really haven’t thought about [how hypospadias will affect me] like when I’m older that much yet. (Participant A) I have no idea [of how I feel about how my penis looks]. (Participant F)
Parent-Proxy Descriptions of Youth Experiences
Setting Expectations	Despite some of his objections at times, when [he] ask[s] me questions, I’m very honest with [him] … as far as exactly what they were going to do [in the surgery] to my knowledge and everything, I mean, we were very open about it. (Parent G)
Surgical Outcomes	I feel like everything is a lot better than it was before. I think that he has more control. (Parent G) [His urine stream] sprays. It kinda, like, goes … different directions instead of just like a stream. (Parent C)
Parent Experiences
Provider Experience	I was lucky enough to have a urologist that I think was, um, very knowledgeable, and not only did he know what he was doing, but I’ve been told by several people that, like, he is one of the best … So, I think that makes me feel a little bit more comfortable that not only was it done right, but that lessens the chance of anything having to be corrected in the future. (Parent G)
Education and Advocacy
• Seeking Support	If there’s any issue, then that’s something that we have to approach together as far as, like, talking to someone and … giving him a more understanding about what’s going on with his body. (Parent I)
• Self-Education	I’ve had other issues I’ve had to do with, that I’ve had to read up more on. (Parent F)
• Provider Education	I probably would have—probably have reconsidered about another route and the reconstruction of the [hypospadias] … because I didn’t know that growth would affect it, you know, the outcome—of the surgery. (Parent I)
Surgical Outcomes
• Penile Function	I feel like, okay, well, that means that it’s … obviously functioning properly, so I don’t really have any concerns. (Parent C)
• Penile Appearance	[T]hey couldn’t come all the way to the end with the opening, but it’s way closer than it was. (Parent B)
• Complications	I actually did bring it up with the, um, last doctor that … it was curved too much … and I did tell him that I was worried about his sexual health when he gets older—and so he actually corrected it. (Parent D)
Parental Trauma from Surgery	… [L]ike I say, during the process of the healing, the pain was just very–very, like, unbear-bearable, you know? And to see him hurt like that was, you know, it was hurtful for me. (Parent I) … [E]very time he opened up, um, a fistula—It was very painful for him … [a]nd it was very traumatic for me. (Parent J)

Theme 1: Penile Factors

Across youth, parent-proxy, and parents, Penile Factors were related to aspects of penile appearance or function, and the interplay of these concerns with the other main themes (psychosocial concerns and expectations of surgery and the healthcare team).

Youth.

Youth focused on experiences related to self-perceptions of penile appearance (satisfaction, dissatisfaction, or uncertainty), penile functions or urinary spraying, and the relationship between their genitalia and bodily changes in early puberty, including erections. Youth also mentioned worry or concern about urinary spraying and penile appearance. Uncertainty, which was centered on worry about post-operative penile appearance and function, encompassed all main themes.

Parent proxies.

Parents discussed their observations of youth bother related to self-comparisons of genitalia to those of close family members or peers. Parents felt that youth dissatisfaction with penile appearance contributed to youth worry or embarrassment. Parents also recalled youth inquiries and conversations about erections and frustrations with urinary control.

Parents.

Parents discussed their child’s penile size and urinary control when considering priorities for their child’s future, and related worries. They endorsed additional frustrations with maintaining toilet hygiene in public restrooms or using restrooms at friends’ homes.

Theme 2: Psychosocial Concerns

Psychosocial Concerns were focused on the burden of anxiety, worry, depression, or interaction with friends, family or peers as related to having hypospadias.

Youth.

Youth described worry related to public exposure in bathroom urinals, and the importance of limiting discussion of surgery to a single parent or very close friends. Traumatic memories from previous surgeries contributed to a fear of future surgeries or persistent worry that hypospadias or complications thereof would persist. While some youth reported active avoidance of disclosure of their condition, others openly discussed surgery with close family or friends. Some also reported romantic interests in peers, which was not affected by or related to penile appearance.

Parent proxies.

Parents discussed youth experiencing embarrassment using public restrooms before surgery, especially related to urinary spraying, while others associated heightened postoperative genital awareness with avoidance of public restrooms. Youth also described fears of future surgeries to parents due to concerns about catheterization or pain. While some youth told parents of discussions of hypospadias with friends or teachers, others limited discussion to a single parent. In discussions with parents, youth romantic relationships were not clearly linked to penile appearance or hypospadias.

Parents.

Parents framed their children’s growth and development through comparison to peers and counseling received before or after their birth. Concerns about judgment or stigma discouraged parents from openly discussing hypospadias with others, prompting them to prioritize shielding their child from public exposure. The anxieties parents faced ranged from worries about future fertility and potential complications overlooked during the child’s increasing independence to concerns about embarrassment stemming from abnormal penile function or appearance. Additionally, parents expressed a desire to ensure their child’s penile appearance adhered to societal norms and their own parental standards (e.g., peer comparisons, parental surgical expectations). Parents expressed apprehensions regarding potential future surgical complications, expressions of sex and gender, and disclosure of their child’s situation to others as they navigated seeking support for their child’s care. Others voiced worries about delayed communication of surgical complications or a fear of overlooking future complications as their youth gained independence, leading to perceived delays in receiving necessary surgery.

Theme 3: Expectations of Surgery and the Health Care Team

Youth and parents described their understanding of what was done during surgery, what to expect in the future, and how surgery and post-surgical complications were experienced by or explained to them.

Youth.

While some youth denied any previous knowledge of their surgery, some described concerns about complications, traumatic memories of surgery, or benefits of surgery related to aiming their urinary stream. A sense of uncertainty about the future lingered for some youth, as well as uncertain impressions related to penile function or appearance.

Parent proxies.

Traumatic perioperative experiences did color youth experiences, as described above. Youth requested parents to detail expectations of what surgery would do and how the surgical site would feel afterward. Youth discussed surgical outcomes with parents in the context of urinary functionality.

Parents.

Parents’ perceptions about physicians’ experience affected parental comfort proceeding with surgery. Education received by parents from providers played a pivotal role in surgical decision making, but parents also cited efforts to find answers online. Some parents reported having to make significant efforts to advocate for their children to be referred to a specialist or to receive care for possible surgical complications. Parent perceptions of surgical success encompassed penile appearance and function, and parents described vigilant observation to ensure that youth did not suffer from untreated complications. Parents endorsed adverse psychological effects of memories of surgical trauma related to overall healing, catheterizations, or development of complications. Parents also discussed heightened worry about urinary control and concerns about finding adequate support for education of their child regarding hypospadias.

Patterns of themes across groups

Mixed methods analyses noted some differences for youth who had surgery after age 2. One older youth cited memories of the catheter or awareness of erections, and two described crushes/attractions. Two parents remembered youth describing urinary control and 3 described youth disclosure to friends or discomfort with disclosure. One parent each cited the importance of setting expectations prior to surgery, needing to seek support, personal avoidance of disclosure, concerns about penile size or delayed communication of complications. Patterns based on ADI (disadvantaged or advantaged neighborhoods), insurance status (public or private), and history of complications (present or absent) are noted below.

Area deprivation indices.

Different aspects of all three main themes varied across ADI groups. Within families from disadvantaged neighborhoods, two youth had asked their parents about erections, but lacked previous understanding of what to expect. Although one denied any knowledge of their prior surgery for hypospadias, four remaining youth with memories expressed fear of future surgeries and memories of trauma, and three youth repudiated any romantic interest in others. Three parents cited a fear of future complications and one family additionally worried about expression of sex and gender. All six parents voiced a fear of disclosure and two cited difficulty seeking support for their child’s care.

In contrast, one youth from an advantaged neighborhoods described knowledge of normal penile functions, including erections. Two parents reported open conversations about urinary control prior to surgery and three reported overall genital awareness after surgery, leading to reluctance to use public urinals for two of these youth. One parent noted that their youth did not experience any adverse effects on their romantic interest in others.

Public versus private insurance.

Three youth with public insurance discussed benefits of surgery and expectations of surgical outcomes, while four discussed traumatic memories of surgery. Four youth noted open discussion of surgery with friends or teachers and two endorsed romantic interest in others. Two parents described youth satisfaction with penile appearance after surgery, but two parents noted youth focus on urinary control as a primary marker of surgical success. One youth asked his parent to have clear conversations about what to expect after repair.

Two parents of youth with public insurance worried about overlooking surgical complications as youth gained independence, while three families noted worries about evolving problems with urinary control and concerns about finding adequate support to educate their child about hypospadias.

One youth with private insurance had no prior knowledge of surgery.

Presence versus absence of complications.

Amongst families who experienced post-operative complications, two parents noted youth asking them to explain how erections work, and three cited parental concerns about future complications. Two youth avoided discussion of hypospadias with others, while three denied romantic interest in others.

Discussion

This qualitative study characterized the experiences of families dealing with hypospadias in the pre-pubertal period, and the three cross-cutting themes (Penile Factors, Psychosocial Concerns, and Expectations of Surgery and the Health Care Team) allowed us to update and streamline our conceptual model to guide HRQOL-focused care. The consistent, higher-level themes and subthemes across youth and parent-proxy experiences (Fig. 1) underscore the value of capturing patient and parent-proxy experiences to monitor HRQOL for youth after repair.

The Penile Factors theme provides qualitative context to prior research which suggested that awareness of penile function changes as patients with hypospadias age [6,7,18–20]. Similarly, findings of a critical literature review, focus groups, and a panel of patient and professional experts included themes of satisfaction with urination and genital appearance and overall well-being for patients and caregivers of youth age 7–17 [7]. Building on these studies, our Penile Factors theme highlights satisfaction with penile appearance and urinary control as a core domain of HRQOL in this population.

The Psychosocial Concerns theme expands on previous findings about the relationship between youth embarrassment, penile appearance, and psychosexual health [21], as well as prior data about parents experiencing anxiety and worry as youth undergo treatment [22]. Extending these previous studies, our findings highlighted a much broader range of psychosocial considerations experienced by parents of pre-pubertal children. For example, youth avoidance of disclosure, the psychological impact of self-comparisons and embarrassment, and parents’ worries about future fertility, complications, psychological health, and normality have not been reported previously. Awareness of these psychological concerns, especially as they relate to penile function, may be central to supporting HRQOL in this population.

The Expectations of Surgery and the Health Care Team theme was not represented in our original conceptual framework [11]. This finding indicates that measures of hypospadias-specific HRQOL should consider parent and youth satisfaction with quality of education from providers and overall communication flows with providers. Previous investigators have discussed the importance of assessing youth and parent satisfaction with penile appearance and penile function (erections and voiding) to define successful outcomes after repair [23]. Effective counseling on this topic requires educating both general pediatric providers and pediatric urologists to use a patient-centered approach to optimize decision-making about hypospadias treatment [24]. Subthemes emphasized the importance of provider experience and setting clear expectations for surgery for youth and parents to decrease parent and youth trauma, uncertainty, and worries about the future.

Sexual health concerns (i.e. erections) were not prominent for our pre-pubertal youth or parents and are likely of greater concern or importance to families in a post-pubertal setting. Recent research on pubertal adolescents indicates youth as young as 16 years old have concerns about sexual function and health requiring treatment [8], and that early surgical intervention can protect psychological adjustment in pubertal youth [25]. Therefore, we recommend continued consideration of sexual health in any measure of hypospadias-specific HRQOL in youth who have progressed into and through puberty.

A unique feature of our study is the inclusion of participants with a diverse range of experiences and backgrounds, which increases the degree to which our themes may be applicable to other populations. Considering these themes in each patient’s clinical and demographic context can help providers personalize patient education and care. Our mixed methods analysis indicates that counseling for families from socioeconomically deprived neighborhoods should directly address normal penile functions, ensure youth and parent understanding of the surgery, address the general fear of open discussion by both youth and parents, and provide strategies for long-term youth support for hypospadias to allay parental concern. For families with public insurance, providers can include more detailed counseling on normal penile functions and monitoring strategies for complications. Similarly, initial provider discussions with families who have had postoperative complications should facilitate open communication of concerns about symptoms with loved ones or providers.

Limitations

We had difficulty finding contact information for many potential study participants, and identified that our patients’ median age at repair was 2 years old, indicating that our sample had later surgery than national averages. These findings suggest an inherent sampling bias, and highlight a need for better infrastructure to maintain contact and provide long-term follow-up care for youth with hypospadias.

Although we reached thematic saturation and adhered to guidelines for qualitative research sampling [26], we did not acquire patient numbers sufficient to draw conclusions about the experiences of different populations or severities of hypospadias. However, we did reach a diverse population of patients in terms of hypospadias severity, race and ethnicity, and socioeconomic status, which allowed us to preliminarily understand the lived experiences of youth and families within a social context.

Conclusion

When assessing hypospadias-specific HRQOL, urologists should consider common HRQOL facets such as psychological considerations, penile factors, and relationships between the healthcare team and family, and employ an individualized approach to counseling and care delivery. Patterns in HRQOL themes across socioeconomic status and insurance coverage suggest potential disparities in access to information and quality care. Our findings from this study will be used to guide expanded studies of patients in different stages of development and facilitate creation of developmentally appropriate PROMs and long-term support tools for people who have been treated for hypospadias. These tools will facilitate holistic care and collaboration between families, behavioral health specialists, and urologists.

Abbreviations

HRQoL

Health-Related Quality of Life

ADI

Area Deprivation Index