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Hypospadias: Adult Sexual Function and Fertility Concerns After Pediatric Repair
By: Megan Stout, MD, The Ohio State University Wexner Medical Center, Columbus; Nicholas Beecroft, MD, The Ohio State University Wexner Medical Center, Columbus; Christina Ching, MD, Nationwide Children’s Hospital, Columbus, Ohio | Posted on: 20 Apr 2023
Hypospadias is one of the most common congenital anomalies in boys, with an estimated prevalence of 1 in 150-300 live births.1 It consists of the classic triad of a dorsally hooded foreskin, an ectopic urethral meatus, and ventral penile curvature. Hypospadias can present in a wide clinical spectrum. With mild presentation, consisting of a meatus located on the glans penis and no chordee present, surgical intervention can be potentially avoided with minimal functional impairment. On the severe end, patients can have debilitating sexual and voiding function that is significantly impaired and, if left uncorrected, can suffer from shorter penile length, painful erections, inability to direct urine stream, and poor body image through adulthood. Traditional classification of hypospadias is based on location of the urethral meatus; utilizing this criterion alone, the majority of boys (70%-85%) have a distal variant, while a small subset have a more severe proximal hypospadias. Other important anatomical considerations consist of degree of penile curvature, degree of glans anomaly, and associated penile shaft skin deficiencies. The anatomical findings present can correlate with future complication development.2
The goals of penile reconstruction in hypospadias repair are to allow the patient to void upright with normal speed/flow, to obtain satisfactory sexual function with a straight penis, and to create an orthotopic meatus with a well-approximated glans for both cosmetic and reproductive motivations. The phenotypic heterogeneity of the condition makes achieving these goals surgically a challenge. Over the past 50 years, there have been advances in hypospadias repair, vastly improving postoperative appearance and function. Techniques such as meatal advancement with glanuloplasty, glans approximation procedures, and tubularized incised plate urethroplasty have all contributed to high success rates in distal repairs. Proximal repairs have historically had less satisfying outcomes, with a high incidence of complications and need for reoperation in adolescence and adulthood.
It is critical to understand the ramifications of childhood hypospadias repair on adult sexual function and fertility potential. The cumulative number of patients having undergone prior hypospadias repair is substantial, and warrants an understanding even in the adult urology practice setting. In regard to future sexual function of this patient population, recent literature suggests that patients with hypospadias have comparable experiences of sexual debut, interest in sex, libido, and satisfaction with sexual experiences comparable to age-matched controls.3 From an initial surgical technique perspective, every effort is made to preserve erectile function: neurovascular bundles are avoided and erectile bodies preserved in ventral lengthening and dorsal plication repairs. On the other hand, the prevalence of erectile dysfunction (ED) is difficult to interpret due to the commonly multifactorial nature of ED predisposition. When considering all hypospadias repairs, multiple studies have shown no difference between patients and controls—though these are limited by small study cohorts.4
Those with a history of proximal hypospadias repair may have a 2-4 times increased risk of mild to moderate ED compared to distal repairs, and an estimated one-third of patients with a history of multiple failed repairs suffer from ED.5 However, there is evidence that the majority of patients may respond to oral pharmacological therapy. Husmann found in a retrospective review of 100 patients with multiple failed hypospadias repairs that 62% of patients with moderate to severe ED responded favorably to an oral phosphodiesterase inhibitor.5 Largely, it seems that patient dissatisfaction as it relates to sexual function appears to stem from the perception of decreased penile length following surgery. Rynja et al reported that patients who underwent proximal repair had significantly shorter mean stretched penile length compared to age-matched controls, while those with distal repairs had no significant difference.6 Perceived decreased penile length was the most influential factor when dissatisfaction in cosmesis and ED was reported. Adult urologists should be aware of patient concerns for decreased penile length and the impact on psychosexual health for appropriate counseling on expectations.
Overall, patients and their partners seem to be far more satisfied with penile appearance than surgeons.4 Surgeons themselves may place a greater emphasis on meatal position than patients do. Andersson et al reported that all patients were “satisfied” or “very satisfied” with meatal position despite only 11% actually having a distal glanular meatus.3 In another study of women asked to rate penile appearance, meatal position or location on the penile shaft was rated as the least important factor.7
Ejaculatory dysfunction can also occur with hypospadias repairs, with an estimated incidence of 12.3% in one study.3 Post-orgasmic milking of the urethra for ejaculate expression may be required of patients with long, reconstructed neourethras. Anejaculation can be related to the reconstructed neourethra, bladder neck incompetence in the individual, or a persistent prostatic utricle.8 A prostatic utricle is an embryological remnant of the mu¨llerian duct; it is found more commonly in boys with proximal hypospadias (estimated 11%-14%). Excision of the remnant can be technically difficult and therefore is reserved for those with refractory bothersome lower urinary tract symptoms, recurrent urinary tract infections, and incomplete emptying rather than ejaculatory dysfunction alone.
In regard to fertility, these patients may have underlying testicular dysfunction with abnormalities in hormone production and/or semen parameters. Thorough evaluation of the patient should include a semen analysis, a physical exam (including Tanner staging, meatal location, testicular size/position, presence of curvature), and a detailed history. In general, adult men following hypospadias repair as a child do not have impaired semen parameters compared to the general population. When stratified by variant severity, those with isolated distal hypospadias have better fertility parameters than those with proximal hypospadias or those associated with other genital disorders.9 Kumar et al found that in men with proximal hypospadias classification, semen analyses had decreased semen volume, sperm concentration, sperm motility, and normal morphology compared to distal variants and controls.9
Several studies have suggested that patients with hypospadias had a lower probability of having a biological child, which was demonstrated in a population-based cohort study of 1.2 million Swedish men with both distal and proximal hypospadias.10 Lower rates of biological children were reported in men with proximal hypospadias compared to those with distal variants or controls. Overall, it is still uncertain whether the decreased paternity rate is a direct result of subfertility and testicular dysgenesis in this population, or related to the concurrent psychosocial factors or body image perception leading to sexual dysfunction.
In conclusion, it is imperative for urologists to be able to identify and diagnose long-term urological complications related to pediatric hypospadias repair. These individuals can present as adult patients in practice with various issues related to sexual and reproductive function in which appropriate evaluation and counseling are imperative. Objective data, validated questionnaires, and consistent long-term follow-up are essential to adequately diagnose these complications that could lead to interventions in adulthood.
- Nelson CP, Park JM, Wan J, Bloom DA, Dunn RL, Wei JT. The increasing incidence of congenital penile anomalies in the United States. J Urol. 2005;174(4 Pt 2):1573-1576.
- Merriman LS, Arlen AM, Broecker BH, Smith EA, Kirsch AJ, Elmore JM. The GMS hypospadias score: assessment of inter-observer reliability and correlation with post-operative complications. J Pediatr Urol. 2013;9(6):707-712.
- Andersson M, Sjöström S, Wängqvist M, Örtqvist L, Nordenskjöld A, Holmdahl G. Psychosocial and sexual outcomes in adolescents following surgery for proximal hypospadias in childhood. J Urol. 2018;200(6):1362-1370.
- Tack LJW, Springer A, Riedl S, et al. Psychosexual outcome, sexual function, and long-term satisfaction of adolescent and young adult men after childhood hypospadias repair. J Sex Med. 2020;17(9):1665-1675.
- Husmann DA. Erectile dysfunction in patients undergoing multiple attempts at hypospadias repair: etiologies and concerns. J Pediatr Urol. 2021;17(2):166.e1-166.e7.
- Rynja SP, Wouters GA, Van Schaijk M, Kok ET, De Jong TP, De Kort LM. Long-term followup of hypospadias: functional and cosmetic results. J Urol. 2009;182(4 Suppl):1736-1743.
- Ruppen-Greeff NK, Weber DM, Gobet R, Landolt MA. What is a good looking penis? How women rate the penile appearance of men with surgically corrected hypospadias. J Sex Med. 2015;12(8):1737-1745.
- Jiao C, Wu R, Xu X, Yu Q. Long-term outcome of penile appearance and sexual function after hypospadias repairs: situation and relation. Int Urol Nephrol. 2011;43(1):47-54.
- Kumar S, Tomar V, Yadav SS, Priyadarshi S, Vyas N, Agarwal N. Fertility potential in adult hypospadias. J Clin Diagn Res. 2016;10(8):PC01-PC5.
- Örtqvist L, Fossum M, Andersson M, et al. Sexuality and fertility in men with hypospadias; improved outcome. Andrology. 2017;5(2):286-293.
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