Hypospadias surgery: understanding parental emotions, decisions and regrets.

This paper builds an argument about genital surgery in the context of medical treatment of children and young people with variations in sex characteristics. First, I set out what is known from existing research including psychological research, surgical follow-up studies and parental regret studies. Second, I present an analysis of surgeons' talk about children, young people and parents in relation to genital surgery. This paper focuses most specifically on hypospadias surgery, but the argumentation is relevant for other kinds of genital surgery carried out in the context of genital variations. The questions guiding this paper are: what research evidence supports hypospadias surgery and what research evidence brings this surgery into question? How might a new interpretation of the evidence, in light of psychosocial research and human rights concerns, contribute to a new perspective on elective genital surgery on minors with variations in sex characteristics? I draw out implications for clinicians supporting parents to decide whether a surgical pathway is the best option for their child.

The hypospadias-specific health-related quality of life conceptual framework: a scoping review of the literature.

PURPOSE: Health-related quality of life (HRQoL) is an important yet understudied construct for individuals with hypospadias (HS). An important barrier towards understanding HRQoL and integrating it into research and clinical care for people with HS is the absence of an underlying conceptual framework to illustrate this construct. We propose a conceptual framework for HS-specific HRQoL based upon a scoping review of the HS literature. METHODS/MATERIALS: We conducted a literature review of articles published between 1989 and 2019. Our search in Embase and Pubmed used the keyword "hypospadias" in combination with "quality of life" and "patient-reported outcomes." We used thematic analysis of the resulting publications to identify core HRQOL domains. From these results and review of HRQoL literature in other pediatric populations, we developed a conceptual framework representing HS-specific HRQoL. RESULTS: We identified five domains of HRQOL previously studied in research with youth and adults with HS: penile appearance, voiding, social interaction, sexual health, and psychological or behavioral function. We propose a model of HS-specific HRQoL comprised of these domains and their areas of overlap, based upon the findings and conceptual mapping of our literature review. CONCLUSION: This novel conceptual framework provides a foundation for understanding disease-specific HRQoL in individuals with HS and may serve as a guide for the conduct of future qualitative studies of the HS population. The overlapping biopsychosocial domains illustrate the possible effects of HS on day-to-day life. This framework may guide future surgical, clinical, and behavioral interventions that aim to improve medical care and quality of life outcomes for HS patients.

Long-Term Outcomes of Distal Hypospadias Repair: A Patients' Point Of View.

PURPOSE: To assess the long-term outcomes of patients treated for distal hypospadias. Assessment of long-term follow-up for a homogeneous population with hypospadias is difficult and there has consequently been a paucity of publications in this regard. MATERIALS AND METHODS: A retrospective review was carried out to compile cases of distal hypospadias operated at our center between 1990 and 1999 according to the MAGPI procedure. Four parameters were evaluated based on four validated questionnaires: Health-related quality of life (SF-36), Genital self-perception (PPS), Self-esteem (Rosenberg Self-Esteem Scale), and Erectile function (IIEF). RESULTS: A total of 77 patients who had undergone MAGPI surgery for hypospadias during the specified period were selected. Sufficient clinical data were available for 51 patients and only 15 of these patients were included, after a median follow-up of 22 years (20-26). Their outcomes were compared with those for a population of 15 matched circumcised men and 15 matched uncircumcised men. No significant difference was found between the patients and the control groups in terms of the score for quality of life (p = .29). There were, however, significant differences in the scores for self-perception of the penile cosmetic appearance (13.3 vs. 15.8; p < .01), self-esteem (30.6 vs. 35.8; p < .01), and erectile function (31.4 vs. 33.7; p = .04) between the patients and the controls. Lower self-esteem correlated with poor genital self-perception (r  = .92). CONCLUSION: This study confirms that adult patients operated for distal hypospadias have poor genital self-perception. This poor genital perception correlated with lower self-esteem.

Coping styles in patients with hypospadias.

OBJECTIVE: The objective of this study is to investigate the coping styles used by patients with hypospadias. Long-term hypospadias studies generally show satisfying outcomes, but some report a lower quality of life (QoL) or poorer psychosocial adjustment, particularly in patients with proximal hypospadias. A lower QoL or psychosocial adjustment was found to be associated with passive coping styles in other pediatric patient populations. Hypothetically, patients with hypospadias also develop different coping styles due to medical experiences in childhood, leading to the lower QoL outcomes on the long term. However, coping styles of patients with hypospadias have never been analyzed. PATIENTS AND METHODS: Adult men with hypospadias repair in childhood were recruited (n = 55; aged 19.9 [IQR 19.2-22.1]). Coping styles were determined with the Utrecht Coping List (UCL) and results compared with a reference group of male students (n = 55, age 20-30 years, no medical history). Sub analysis of coping styles of the hypospadias groups was done based on three items: severity of hypospadias, time of last hypospadias surgery and occurrence of postoperative complications. RESULTS: Compared to the reference groups, patients with hypospadias had higher scores on Avoidance (P < 0.05), particularly patients who had ≥1 postoperative complication or proximal hypospadias. Patients with proximal hypospadias also had lower scores on Seeking Social Support compared to the reference group (P < 0.05). Within the hypospadias group, coping style scores did not differ based on severity of hypospadias, timing of surgery or postoperative complications. CONCLUSION: Patients with hypospadias, particularly those with proximal hypospadias or a postoperative complication, more often use an avoiding coping style compared to a reference population. Attention to coping styles during the follow-up of patients with hypospadias might help to improve the QoL in these patients.

Parental perspectives on decision-making about hypospadias surgery.

INTRODUCTION: Many parents who choose hypospadias repair for their son experience decisional conflict and regret. The utilization of a shared decision-making process may address the issue of decisional conflict and regret in hypospadias repair by engaging both parents and physicians in decision-making. OBJECTIVE: The objective of this study was to develop a theoretical framework of the parental decision-making process about hypospadias surgery to inform the development of a decision aid. STUDY DESIGN: We conducted semistructured interviews were conducted with parents of children with hypospadias to explore their role as proxy decision-makers, inquiring about their emotions/concerns, informational needs, and external/internal influences. Interviews were conducted until no new themes were identified, analyzing them iteratively using open, axial, and selective coding. The iterative approach entails a cyclical process of conducting interviews and analyzing transcripts while the data collection process is ongoing. This allows the researcher to make adjustments to the interview guide as necessary based on preliminary data analysis in order to explore themes that emerge from early interviews with parents. Grounded theory methods were used to develop an explanation of the surgical decision-making process. RESULTS: Sixteen mothers and one father of seven preoperative and nine postoperative patients (n = 16) with distal (8) and proximal (8) meatal locations were interviewed. Four stages of the surgical decision-making process were identified: (1) processing the diagnosis, (2) synthesizing information, (3) processing emotions and concerns, and (4) finalizing the decision (Extended Summary Figure). Core concepts in each stage of the decision-making process were identified. Primary concerns included anxiety/fear about the child not waking up from anesthesia and their inability to be present in the operating room. Parents incorporated information from the Internet, medical providers, and their social network as they sought to relieve confusion and anxiety while building trust/confidence in their child's surgeon. DISCUSSION: The findings of this study contribute to our understanding of decision-making about hypospadias surgery as a complex and multifaceted process. The overall small sample size is typical and expected for qualitative research studies. The primary limitation of the study, however, is the underrepresentation of fathers, minorities, and same-sex couples. CONCLUSIONS: This study provides an initial framework of the parental decision-making process for hypospadias surgery that will inform the development of a decision aid. Future stages of decision aid development will focus on recruitment of fathers, minorities, and same-sex couples in order to enrich the perspectives of our work.

Community engagement of adolescents in the development of a patient-centered outcome tool for adolescents with a history of hypospadias repair.

INTRODUCTION: Hypospadias may lead to long-term issues with urination, sexual function and psychosocial well-being. Limited evidence exists regarding the healthcare communication preferences of male adolescents regarding sensitive topics. OBJECTIVE: The purpose of this qualitative study was to explore the healthcare communication preferences of male adolescents regarding sensitive topics (e.g. urinary and sexual issues) and engage them in the initial stages of development of a patient-centered outcome tool for adolescents with a history of hypospadias repair. STUDY DESIGN: A multidisciplinary team with communication design expertise, pediatric urology experts, and health services researchers developed a self-reported toolkit for adolescent patients who had hypospadias repair as children. The toolkit featured short writing/diagramming exercises and scales to facilitate participant reflections about genital appearance, urination, sexual function, and psychosocial well-being. We recruited students from two local high schools for two focus groups to obtain feedback about the usability/acceptability of the toolkit's appearance/content. We inquired about language preferences and preferred format and/or setting for sharing sensitive information with researchers. The focus groups were audio recorded, professionally transcribed, checked for accuracy, and analyzed by two coders using qualitative content analysis. Major themes and subthemes were identified, and representative quotes were selected. RESULTS: We conducted two focus groups in January 2018 with 33 participants, aged 14-18 years. Participants preferred language that would make patients feel comfortable and serious, clinical language rather than slang terms/sexual humor (Extended Summary Table). They recommended avoidance of statements implying that something is wrong with a patient or statements that would pressure the patient into providing answers. They suggested fill-in-the-blank and open-ended responses to encourage freedom of expression and colorful graphics to de-emphasize the test-like appearance of the toolkit. Most participants preferred a toolkit format to a one-on-one interview to discuss sensitive topics such as urinary or sexual issues. Participants would prefer either a male interviewer or would like to have a choice of interviewer gender for individual qualitative interviews, and they recommended a focus group leader with a history of hypospadias repair. DISCUSSION: This study provides a rich description of a group of male high school students' experiences with healthcare providers and researchers. Its qualitative design limits generalizability, and our findings may not be similar to those of adolescents with a history of hypospadias repair. CONCLUSION: We used focus group feedback on the toolkit prototype to refine the tool for use in a future study of adolescents with a history of hypospadias repair.

No difference in cognitive performance or gender role behavior between men with and without hypospadias.

BACKGROUND: Hypospadias is a common malformation of the male external genitalia that results in urethral displacement with different levels of severity. Male genital development during the fetal period is dependent on androgen function, while the etiology of hypospadias differs and can be multifactorial. The psychosocial outcome is sometimes affected, but according to several studies acceptable. The question of whether hypospadias is associated with differences in psychosexual development has been investigated previously, with mixed results. There are no previous investigations of cognitive abilities in men with hypospadias. OBJECTIVE: The aim of this study was to investigate whether hypospadias is associated with differences in performance on cognitive tests and/or gender role behavior. PARTICIPANTS: Eighty-six men with hypospadias were compared to male and female controls from the general population. PROCEDURE: Cognitive tasks, previously shown to yield group level sex differences and questions regarding self-reported childhood gender role behavior, were administered either at an outpatient clinic visit or via online participation. RESULTS: The cognitive performance of men and women in the control groups differed significantly in the expected directions. Men and women also differed on self-reported childhood gender role behavior questions. There were no significant differences between men with and without hypospadias on any of the measures. Men with proximal hypospadias performed slightly lower on many of the cognitive tasks in comparison to men with distal hypospadias and controls. CONCLUSION: In general, hypospadias is not associated with differences in performance on cognitive tests that typically yield sex differences or with altered gender role behavior in childhood. Further studies on cognitive abilities in boys and men with proximal hypospadias are warranted.

Psychiatric symptoms in men with hypospadias - preliminary results of a cross-sectional cohort study.

AIM: Population studies have shown an increased risk of neurodevelopmental disorders in males born with the congenital condition hypospadias, where the opening of the urethra is on the underside of the penis. We investigated overall psychiatric morbidity in cases and matched controls. METHODS: This study compared 167 men born with hypospadias from 1959 to 1994 in Stockholm or Gothenburg in Sweden using hospital registers. They were compared with controls from the Swedish population registry, who were contacted by regular mail and students who were recruited by local advertisements. The total sample had a mean age of 33.5 years (range: 19-54). They completed self-rating scales for depressive, anxiety and obsessive-compulsive symptoms and symptoms of attention deficit hyperactivity disorder. In addition, 33 cases and 47 controls underwent psychiatric morbidity interviews that covered the 17 most common psychiatric diagnoses. RESULTS: A fifth (21%) of both the cases and controls reported current or previous psychiatric symptoms. There were no significant differences in self-rated depression, anxiety or obsessive-compulsive disorder symptoms between the patients and controls or between the different phenotype groups. The distribution was not significantly affected by the severity of hypospadias. CONCLUSION: Psychiatric morbidity was no higher in men with hypospadias than population-based controls.

Postoperative behavioral changes in Chinese children undergoing hypospadias repair surgery: A prospective cohort study.

BACKGROUND: The perioperative period can be psychologically challenging, and children may exhibit behavioral changes following surgical anesthesia. It is unknown whether children in China have additional risk factors associated with negative behavioral changes. OBJECTIVES: The aim of this study was to investigate the incidence of behavioral changes in children after hypospadias repair surgery and to identify potential risk factors associated with negative behavioral changes. METHODS: A prospective cohort of 177 children aged 2-12 years scheduled for hypospadias repair surgery from 2016 to 2017 was studied. The primary outcome was the incidence of behavioral changes on postoperative days 14 and 30 evaluated with the Post-Hospitalization Behavioral Questionnaire. Data collected included demographic data, anesthesia details, procedure details, admission details, child anxiety, child temperament, pain, and emergence delirium. Multivariable logistic regression was used to identify risk factors associated with postoperative negative behavioral changes. RESULTS: A total of 60.5% (107/177) of children exhibited negative postoperative behavioral changes on day 14 and 46.5% (79/170) exhibited changes on day 30 after the surgery. Approximately 2.3% (4/177) and 2.4% (4/170) of children showed improved behavior on postoperative days 14 and 30, respectively. The frequency of temper tantrums changed the most. The logistic regression results suggested that a younger age (odds ratio: 0.86; 95% confidence interval 0.76-0.96), emotional temperament (odds ratio: 1.1; 95% confidence interval 1.0-1.2) and maternal education (odds ratio: 2.2; 95% confidence interval 1.1-4.5) were associated with negative postoperative behavioral changes on day 14. On day 30, a younger age (odds ratio: 0.87; 95% confidence interval 0.77 to 0.98) was the only factor associated with negative postoperative behavioral changes. CONCLUSION: For children undergoing hypospadias repair surgery in our institution, approximately three in five showed postoperative behavioral changes. In addition to a younger age and a higher maternal education, an emotional temperament is associated with a high incidence of negative postoperative behavioral changes.

Gender identity in patients with 5-alpha reductase deficiency raised as females.

BACKGROUND AND OBJECTIVE: 5-Alpha reductase type 2 deficiency (5-ARD) is a rare disorder of sex development. The lack of 5-alpha reductase, an enzyme that converts testosterone into dihydrotestosterone, results in external genitalia that may appear female, or predominantly male, albeit undervirilized, or, more often, ambiguous. METHODS: This study describes a series of patients with 5-ARD raised as female, focusing on aspects related to gender identity. Following a retrospective chart review, patients with 5-ARD were invited to return to the clinic to enable their gender identity to be assessed using an 11-item structured in-house questionnaire. The Golombok-Rust Inventory of Sexual Satisfaction was applied to patients who had initiated their sexual life. RESULTS: Six patients aged >15 years with 5-ARD and raised as female were included. Most patients were diagnosed late: two before and four after puberty. The mean length of the phallus was 2.8 cm (0.5-5.0). Reasons for seeing a doctor included genital appearance (n = 3), amenorrhea/absence of breast development (n = 2), and changes in gender role attitudes (n = 1). According to the gender identity assessment, 4 patients identified as female, 1 as male, and 1 as both genders. Only the patient identified as male requested gender re-assignment. Of the two patients who had initiated their sexual life, sexual satisfaction was found to be good in one and poor in the other due to vaginal discomfort during intercourse. CONCLUSION: In the present series, the majority of undervirilized patients with a diagnosis of 5-ARD raised as female were in complete conformation with being female and described themselves as heterosexual. The more virilized patients were those least in conformity with their female-assigned gender.

Shaping parents, shaping penises: How medical teams frame parents' decisions in response to hypospadias.

INTRODUCTION: Psychological research provides insights into how parents approach medical decisions on behalf of children. The medical decision of concern here is the surgical alteration of a hypospadic penis, whose urethral opening does not appear at the tip. Hypospadias surgery is routinely carried out in infancy, despite criticism by international organizations concerned about children's rights. The focus of this study is on the framing of hypospadias surgery. OBJECTIVES: The objective was to examine how health professionals frame hypospadias and hypospadias surgery in medical and non-medical ways. DESIGN: This was a qualitative study designed to build on the experimental research of Streuli et al. who investigated how medical versus non-medical information affects decision-making about non-essential childhood genital surgery. METHODS: Semi-structured interviews were undertaken with 32 health professionals. Theoretically informed thematic analysis was used to examine how health professionals talk about hypospadias surgery and about supporting parents to make treatment decisions. RESULTS: The analysis suggests that medical professionals' engagement with parents underestimates the effect of framing in influencing parental decisions about hypospadias surgery. Some psychological specialists in this area are actively framing hypospadias in ways that enable some parents to choose a non-medical pathway. Psychologically informed ways of talking about a child's genital difference focus on psychological qualities, including affect, well-being, and unconditional positive regard. CONCLUSIONS: The best interests of children with hypospadias may well be served when psychological pathways are highlighted, providing opportunities to support the flourishing of children whose genital appearance raises the question of medical intervention. Statement of Contribution What is already known on this subject? Framing significantly affects medical decision-making in ways that people typically fail to perceive. Parents frequently consent to non-essential hypospadias (penile) surgery for their sons, despite the risks and ethical concerns. What does this study add? Medical teams could do more to consider framing when counselling parents about their son's hypospadias. Psychological specialists can help parents to frame their son's penile difference in terms of well-being and love. The best interests of children with hypospadias may be served by highlighting psychological care pathways.

Psychosocial and Sexual Outcomes in Adolescents following Surgery for Proximal Hypospadias in Childhood.

PURPOSE: We evaluated psychosocial outcomes, psychosexual development and sexual function in adolescents who had undergone surgery for proximal hypospadias. We hypothesized that these outcomes would be impaired compared to peers. MATERIALS AND METHODS: We identified 55 males age 14 years or older who underwent surgery for penoscrotal to perineal (intraoperatively defined) hypospadias between 1996 and 2005. A total of 33 patients with a median age of 17.5 years (range 14 to 25) answered a Web based questionnaire with self-constructed questions, completed the validated Psychological General Well-Being Index, Body-Esteem Scale for Adolescents and Adults and Penile Perception Score, and underwent clinical evaluation. A total of 31 patients with distal hypospadias (median age 19 years, range 14 to 35) and 25 age matched healthy men (17.5 years, range 14 to 25) served as controls. RESULTS: Interest in sex, age at sexarche and satisfaction with sexual experiences were comparable between patients and controls. Three patients with proximal hypospadias (10%) and 1 control (4%) reported occasional erectile problems. Three patients with proximal hypospadias (11%), 1 patient with distal hypospadias (3%) and 1 control (4%) affirmed anejaculation. There were no differences in results between validated questionnaires. Patients with proximal hypospadias were more dissatisfied with penile length (39%) compared to controls (12%, p = 0.049). Concerning physical contact, 10 patients (38%) expressed uncertainty. Extra support in school was more frequent among patients with proximal hypospadias (p = 0.024 vs distal hypospadias, p = 0.068 vs control group). CONCLUSIONS: Despite concerns regarding penile length, sexual experiences were comparable to those of other adolescents, although more than a third of patients with proximal hypospadias demonstrated uncertainty on questions relating to desire for physical contact. Specialized tutoring in school was more common in patients with proximal hypospadias. Continuous followup throughout childhood allowing extra time for age adequate information and support is warranted.

Steroid 5α-reductase 2 deficiency leads to reduced dominance-related and impulse-control behaviors.

The enzyme steroid 5α-reductase 2 (5αR2) catalyzes the conversion of testosterone into the potent androgen 5α-dihydrotestosterone. Previous investigations showed that 5αR2 is expressed in key brain areas for emotional and socio-affective reactivity, yet the role of this enzyme in behavioral regulation remains mostly unknown. Here, we profiled the behavioral characteristics of 5αR2 heterozygous (HZ) and knockout (KO) mice, as compared with their wild-type (WT) littermates. While male 5αR2 KO mice displayed no overt alterations in motoric, sensory, information-processing and anxiety-related behaviors, they exhibited deficits in neurobehavioral correlates of dominance (including aggression against intruders, mating, and tube dominance) as well as novelty-seeking and risk-taking responses. Furthermore, male 5αR2 KO mice exhibited reduced D2-like dopamine receptor binding in the shell of the nucleus accumbens - a well-recognized molecular signature of social dominance. Collectively, these results suggest that 5αR2 is involved in the establishment of social dominance and its behavioral manifestations. Further studies are warranted to understand how the metabolic actions of 5αR2 on steroid profile may be implicated in social ranking, impulse control, and the modulation of dopamine receptor expression in the nucleus accumbens.

Hypospadias in male infants - a review.

The external genitalia problems are cumbersome problems for both doctors and parents, as these abnormalities have a consequent impact on future generations. However, the affected young infants are unaware of the consequences due to immature emotional state. Further, the feeling of being different and inferior in affected young patients could give rise to negative emotions including depression, insecurity, anxiety, powerlessness, etc. These all factors collectively could cause a mental imbalance in the affected children. The present review article is focused on the latest updates in the area.

Resultados a largo plazo de la cirugía de hipospadias. Función urinaria, sexual e impacto psicológico / Hypospadias: results and psychological alterations

Objetivos. Conocer el estado actual de los pacientes intervenidos por hipospadias en nuestra región, para adaptar la técnica quirúrgica a sus necesidades reales. Material y métodos. Estudio descriptivo y observacional. Se entrevistó a pacientes intervenidos de hipospadias en nuestro hospital entre 1976 y 1996, incidiendo en su función urinaria, sexual e impacto psi- cológico. Realizamos análisis estadístico descriptivo y comparaciones entre tipos de hipospadias (SPSSv19). Resultados. 566 pacientes fueron intervenidos de hipospadias en el período estudiado, entrevistándose a 100, con edades entre 18 y 40 años. El 73% fueron distales y 27% proximales. En el 30% el meato no es ortotópico, 10% presenta fístula, 18% presenta algún grado de estenosis y 52% presenta curvatura. El 13% siente haber tenido desventajas en su vida. Consideran su pene diferente en: tamaño (19%), forma glande (17%), curvatura (14%), cicatrices (10%) y orificio más bajo (7%). La satisfacción sexual fue valorada en 8,9 en una escala del 1 al 10, sin diferencias entre los tipos de hipospadias. Los proximales presentan mayores problemas de eyaculación: 42% frente al 11% de los distales. El 68% de los proximales desearían mejorar frente al 20% de los distales, en apariencia (17%), tamaño (11%) y forma de orinar (11%). Conclusiones. Se mantienen alteraciones anatómicas pero los aspectos que más valora el paciente adulto difieren de los perseguidos en las cirugías que se practicaban. Un porcentaje importante de pacientes se reoperaría de nuevo. Mantener el tamaño del pene y aspecto natural del glande debe ser un objetivo prioritario durante la cirugía de hipospadias (AU)

Objectives. To determine the current status of the patients operated on for hypospadias in our region, in order to adapt the surgical technique to their real needs. Material and methods. A descriptive and observational study. Interviews were conducted with hypospadias surgery patients in our hospital between 1976 and 1996, focusing on their urinary and sexual function and psychological impact. We performed a descriptive statistical analysis and comparisons between types of hypospadias (SPSSv19). Results. 566 patients were operated on for hypospadias during the study period, interviewing 100 aged between 18 and 40 years. The 73% were distal and 27% proximal. Of all patients, 30% do not have an orthotopic meatus, 10% have fistula, 18% have some degree of stenosis and 52% have penile curvature. The 13% feel to have had disadvantages in their life. They consider they have a different penis in: size (19%), glans shape (17%), curvature (14%), scars (10%) and lowest hole (7%). Sexual satisfaction was valued at 8.9 on a scale of 1 to 10, with no differences between the types of hypospadias. Proximal hypospadias patients have more ejaculation problems: 42% versus 11% in distal hypospadias. The 68% of the proximal ones would like to improve compared to 20% of the distal in: appearance (17%), size (11%) and way to urinate (11%). Conclusions. Anatomic abnormalities are maintained but the aspects that values the adult patient differ from those persecuted by practiced surgeries. A significant percentage of patients would try to get another surgery. Keeping the size of the penis and natural appearance of the glans should be a priority in hypospadias surgery (AU)

Update on the surgical approach for reconstruction of the male genitalia.

The majority of patients with DSD will be found to carry an XY karyotype and be assigned male gender. From a phenotypical standpoint, most will present with proximal hypospadias ± cryptorchidism. In this review article, the authors present the current status of reconstruction of the male genitalia in this setting. The report addresses the following topics: surgical input in the evaluation of the newborn with an undervirilized external genitalia, including gender assignment considerations; controversies surrounding timing and indication for hypospadias surgery in proximal cases as well as use of testosterone; surgical techniques and decision-making process for one- vs. two-stage repairs; complications of hypospadias surgery based on technique used for repair; and long-term follow-up. The high complication rates observed in the treatment of proximal hypospadias attest to its challenging nature. Concentration of experience, tracking carefully identified patient-centered outcomes and long-term follow-up of this patient population are recommended.

Maternal Stressors and Social Support and Risks of Delivering Babies With Gastroschisis or Hypospadias.

We examined the association of maternal stressful life events and social support with risks of gastroschisis and hypospadias, using data from the National Birth Defects Prevention Study, a population-based case-control study of US births taking place in 2006-2011. We examined maternal self-reports of 7 life events and 3 sources of social support during the periconceptional period among mothers of 593 gastroschisis cases, 1,142 male hypospadias cases, and 4,399 nonmalformed controls. Responses to the questions on stressful life events were summed to form an index (higher is worse), as were responses to questions on social support (higher is better). We used logistic regression to estimate adjusted odds ratios and 95% confidence intervals. The adjusted odds ratios for gastroschisis for a 4-point increase in the stress index were 3.5 (95% confidence interval (CI): 2.6, 4.8) among nonteenage mothers (age ≥20 years) and 1.0 (95% CI: 0.5, 1.7) among teenage mothers (age <20 years). The odds ratio for hypospadias (among all mothers) was 0.8 (95% CI: 0.7, 1.1). Adjusted odds ratios for a social support score of 3 (versus 0) in the 3 respective groups were 0.6 (95% CI: 0.4, 1.0), 1.0 (95% CI: 0.5, 2.3), and 0.6 (95% CI: 0.4, 0.9). Given the lack of prior research on these outcomes and stress, results should be interpreted with caution.

Psychosocial outcomes in adult men born with hypospadias: A register-based study.

In this nationwide matched cohort study, we have investigated whether being born with hypospadias affect subsequent psychosocial outcomes in adulthood. We analyzed prospectively collected data from national Swedish registers. Data on the diagnoses were collected from the National Patient Register and the Medical Birth Register. Data on psychosocial outcomes such as educational and income level, marital status and disability pension were collected from Statistics Sweden. The effects of covariates, such as age, county of birth, presence of other malformations and psychiatric illness, were taken into account. The associations between hypospadias and psychosocial outcomes were calculated using conditional logistic regression and expressed as odds ratios (OR) and 95% confidence intervals (CI). We included 4378 men diagnosed with hypospadias, born between 1969 and 1993 in Sweden. Patients with hypospadias were matched with unaffected men by year of birth and birth county. We did not detect any differences in educational or income level. The probability of entering marriage (OR 1.02, 95% CI 0.90-1.14) did not differ, regardless of phenotype. We did, however, detect a 40% increased probability of receiving a disability pension, (OR 1.39, 95% CI 1.20-1.61). In conclusion, men born with hypospadias in Sweden do not differ from unaffected men with respect to the majority of psychosocial outcomes studied. They are, however, at increased risk of receiving a disability pension, which motivates further investigations.

Psychosocial outcome in adult men born with hypospadias.

INTRODUCTION: Hypospadias, which is a surgically treated congenital malformation of the male urethra, may have a negative impact on quality of life. This aspect has previously been subject to limited research. This study examined the long-term psychosocial outcome of a large cohort of adult males born with hypospadias. OBJECTIVE: The purpose of this case-control study was to assess a possible negative influence on the psychosocial outcome in adult males with hypospadias. STUDY DESIGN: Males with hypospadias treated in Sweden and aged ≥18 years old participated in this follow-up study. Age-matched men and university students were recruited as controls. The participants answered a questionnaire designed to reflect the subjective quality of life, social factors, need of support and follow-up, and the perceived impact of the disease upon upbringing. It also looked at the validated Psychological General Well-Being (PGWB) questionnaire and Relationship Questionnaire (RQ). RESULTS AND DISCUSSION: A total of 167 patients (median age 34 years, 63% distal, 24% mid, and 13% proximal hypospadias) and 169 controls (median age 33 years) participated in the study. Patients had their first operation at 4 years of age (median) and the median follow-up time was 29 years following the first surgery. Men with hypospadias had a comparable total quality of life level with a mean total PGWB score of 82 (normal range 78-83) compared with 85.6 in controls. Scores on wellbeing and vitality were lower, even if the differences were small. Hypospadias did not affect marital status, presence of children in the family, frequency of employment or experience of bullying. These men more often lived at home with their parents (P=0.001) and had a lower level of education (P=0.004), even if the educational level in both patients and controls was high compared with the general Swedish population. Patients with proximal hypospadias were shorter compared with controls (P=0.003), which was consistent with the prenatal growth restriction associated with hypospadias. The group with proximal hypospadias expressed a greater need for medical (45.5%) follow-up compared with mid (28.2%) and distal (18.1%) cases (P=0.001). Patients with proximal hypospadias tended to avoid close relationships because of fear of being hurt. CONCLUSIONS: The findings suggested that patients treated for hypospadias have a good HRQoL, can be expected to have a normal psychosocial life, and marry and have children. Repeated follow-up and psychological support during childhood/adolescence is however of great importance for patients with more proximal hypospadias.