Long-Term Outcomes of Distal Hypospadias Repair: A Patients’ Point Of View Alexis Belgacem1, Laurent Fourcade1, Romain Pelette2, Etienne Bouchet2, Victor Lescure2, Walter Bertherat2, Grazia Spampinato1, Jean-Luc Alain1, Aurélien Descazeaud2, Quentin Ballouhey1* 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-percep- tion. This poor genital perception correlated with lower self-esteem. Keywords: hypospadias; MAGPI; long-term outcomes; self-esteem INTRODUCTION Hypospadias exhibits a broad spectrum of severity, and a multitude of surgical reconstructive tech- niques have been devised that are aimed at correcting this congenital abnormality. Even for the specific distal type of hypospadias, the large variety of procedures re- flects the absence of an established gold standard for treatment. Irrespective of the choice of the technique used, there is not a clear consensus regarding the in- dication for surgery in case of very distal hypospadias due to the substantial cosmetic considerations involved. There is a paucity of published data regarding the cos- metic and self-estimated outcomes of hypospadias. Most of the published series to date have been in regard to short-term cosmetic outcomes reflecting the opinions of the parents and the surgeon. Definitive urinary func- tional outcomes can be addressed in adolescence.(1) It is, however, more difficult to obtain data regarding sexual function and self-estimated cosmetic outcomes, as this requires long-term close follow-up.(2) There have been few reports to date of long-term outcomes, particularly in regard to distal hypospadias.(3) Similar long-term cos- 1Service de chirurgie pédiatrique, Hôpital Mère-Enfant, Centre Hospitalier Universitaire de Limoges, 8 Avenue Dominique Larrey, 87042 Limoges, France. 2Service de chirurgie Urologique, Hôpital Dupuytren, Centre Hospitalier Universitaire de Limoges, 2 Avenue Martin Luther King, 87000 Limoges, France. *Correspondence: Service de chirurgie pédiatrique, Hôpital Mère-Enfant, Centre Hospitalier Universitaire de Limoges, 8 Avenue Dominique Larrey 87042 Limoges, France Tel: +33/6 32 85 96 60; Fax: +33/5 55 55 86 82, Email: q.ballouhey@gmail.com Received March 2020 & Accepted September 2020 metic and functional issues can be expected for operat- ed patients and patients without hypospadias.(2) The rationale to operate distal hypospadias during early childhood should take into account cosmetic and ethical considerations. The fundamental question is whether esthetic correction is indicated before the patient has the capacity to provide their informed consent4. There has been no large-scale study to date comparing the long- term outcomes of adult patients who have or who have not been operated for distal hypospadias. The key issue remains the self-perception of patients regarding their hypospadias. Most of the data published to date has been in regard to medium-term follow-up series with heterogeneous types of hypospadias and follow-up du- rations.(5,6) Of note, there has been one study that report- ed a similar health-related quality of life for operated patients and controls.(4) No study, however, has focused on the self-estimated outcomes for adult patients oper- ated for distal hypospadias during childhood. To address these issues, we focused on a particular pop- ulation: patients with distal hypospadias operated with a single repair technique. The primary endpoint of the PEDIATRIC UROLOGY Urology Journal/Vol 18 No. 5/ September-October 2021/ pp. 537-542. [DOI: 10.22037/uj.v16i7.6172] present study was to report the long-term outcomes for such patients in terms of the functional parameters, ef- fects on sexual performance, and health-related quality of life (HRQoL) after reconstructive surgery for distal hypospadias with long-term (> 20 years) follow-up. The secondary endpoint was to compare these param- eters with those of the general population without hy- pospadias. METHODS Study population The patients had to be at least 20 years of age to be con- sidered eligible for this study. A retrospective review was carried out to compile cases of hypospadias operat- ed between 1990 and 1999 for distal hypospadias. This study was approved in 2019 by the ethics committee of the University Hospital of Limoges as reference num- ber 309-2019-75. Inclusion and exclusion criteria The primary endpoint was the long-term outcomes of patients treated for distal hypospadias in terms of func- tional outcomes, effects on sexual performance, and general quality of life. A total of 138 medical files of patients who had a procedure code compatible with hy- pospadias repair at our institute between 1 January 1990 and 31 January 1999 were reviewed in order to select pa- tients who were between 20 and 30 years of age in 2019 and who had undergone primary hypospadias repair. Only patients with distal hypospadias repair according to the Meatal Advancement and Glanuloplasty (MAG- PI) procedure were included in the present study. We selected these patients to obtain a homogeneous popu- Long-term outcomes of hypospadias-Belgacem et al. Variables (min-max)a Total Group 1- circumcised Group 2- uncircumcised P-value Number 30 15 15 Age 28.0 (21-39) 28.8 (21-38) 27.2 (21-39) .48 Age at surgery (months) 42.4 (13-65) 42.4 (13-65) none Penile score (PPPS) 15.8 (13-18) 16.5 (15-18) 15.2 (13-18) .07 - Length 2.2 (2-3) 2.4 (2-3) 2.2 (2-3) - Meatus - 2.9 (2-3) - 3.0 (2-3) - 2.9 (2-3) - Glans - 2.6 (2-3) - 2.8 (2-3) - 2.6 (2-3) - Skin - 2.2 (2-3) - 2.8 (2-3) - 2.2 (2-3) - Axis - 2.6 (2-3) - 2.8 (2-3) - 2.6 (2-3) - Appearance - 2.2 (2-3) - 2.4 (2-3) - 2.2 (2-3) Erectile function (IIEF) 33.7 (30-35) 33.8 (33-35) 33.6 (30-35) .44 Self-esteem (Rosenberg) 35.8 (28-40) 36.5 (28-39) 34.9 (28-40) .19 Quality of life (Euroqol5D) 91.3 (65-100) 92.1 (75-100) 90.3 (65-100) .56 Table 1. Characteristics of the control group comprising unoperated and circumcised men Abbreviations: PPPS: Pediatric Penile Perception Score; IIEF: International Index of Erectile Function a The continuous variables were compared using a parametric test (Student’s t-test) and non-a parametric test (Mann-Whitney test) Figure 1. Study flow diagram. Pediatric Urology 538 lation with high expectations regarding the cosmetic re- sults. Distal hypospadias was defined as a preoperative meatal position distal to and including mid-penile shaft cases. These patients did not have preoperative penile curvature. Out of a total of 104 patients, 23 underwent tubularized incised plate repair, 4 underwent Mathieu repair, and 77 underwent MAGPI procedures. The only exclusion criterion was insufficient data regarding short-term follow-up of the patients. During the follow-up, the patients were asked to com- plete a questionnaire regarding their genital self-per- ception. If they were no longer included in close fol- low-up, they were also invited to consult with the dedicated urology unit of our institute. The control group comprised men without hypospadias who agreed to anonymously complete the same questionnaire. Each patient was matched according to age at the time of the surgery and age in 2019 to two adult controls without hypospadias from both a circumcised and an uncircum- cised patient population. The circumcised group was compared to the uncircumcised group, and the hypo- spadias group was compared to the total control group. Four parameters were evaluated based on four validated questionnaires. Health-related quality of life (HRQoL) The 36-item Medical Outcomes Study Short-Form questionnaire (SF-36) was used to assess the HRQoL.(7) This scale has been validated and has been determined to be reliable for our population (reliability coefficient of 0.75).(8) Subscales of each domain (physical function, role limitations, bodily pain, general health perception, mental health, role limitations due to emotional prob- lems, vitality, and social function) were scored from 0 to 100, with higher scores indicating a better HRQoL. Genital self-perception (PPS) Penile cosmetic self-perception was evaluated accord- ing to the Penile Perception Score.(9) This instrument consists of the following four items for the participants to evaluate their genitals, with a 4-point scale ranging from very dissatisfied (0) to very satisfied (3): the posi- tion and shape of the meatus, the shape of the glans, the shape of the shaft skin, and the general appearance of the penis. The PPS score ranges from 0 to 12. It is a rec- ognized score in the domain of hypospadias, although it has yet to be shown to be a validated patient-report- ed outcome instrument.(10) We also assessed two addi- tional items included in the pediatric PPS because their absence is a limitation according to the literature: the length and the axis of the penis.(2, 11) Self-esteem The Rosenberg Self-Esteem Scale, which is a widely used self-report instrument for disorders such as sexual development, was chosen to evaluate the participants’ level of self-esteem.(12) Self-esteem is tightly linked to happiness.(13) This 10-item scale measures overall self-worth by assessment of both positive and negative feelings about oneself. All of the items are rated using a 4-point Likert scale ranging from ‘strongly agree’ to ‘strongly disagree’. Erectile function and orgasmic function The international index of erectile function was used to measure erectile and orgasmic functions in the past 6 months. The erectile function subscale has a maximum domain score of 30 and an organic function subscale of 10.(14) Higher scores correspond to better functions. This scoring method was validated many years ago and it is commonly used in publications relating to studies of hypospadias.(3,14) Statistical analysis The data were analyzed using Prism 8.2.1® Mac® software (GraphPad Software, San Diego, California, USA). The comparative statistics were performed using Fisher’s exact test for the categorical variables, a para- metric test (Student’s t-test) for the data with a normal distribution (means and the corresponding SD are pre- sented), and a non-parametric test (Mann-Whitney test) for the data without a normal distribution (medians and the IQR are presented). A p-value of less than 0.05 was considered significant. RESULTS A total of 77 patients operated for distal hypospadias in the specified period were selected. No postoperative complications occurred after a median follow-up of 15 months (range 1-63). No cases involving penile curva- ture or a short penis length were noted. Clinical data were available for 51 patients to calculate the postoper- ative PPS score. The patients were contacted using their parents’ addresses or through social networks. The cor- rect contact details and addresses of the patients were ultimately only available for 45 of the patients. A total of twenty-nine patients could be reached by phone, of whom twenty-two were lost to follow-up (Figure 1). No Variables (min-max)a Patients Control group P-value Number 15 30 Age 24.3 (20-30) 28.0 (21-39) .02 Age at surgery (months) 42.3 (12-78) 42.4 (13-65) (circumcision) .86 Penile score (PPPS) 13.3 (10-17) 15.8 (13-18) < .001 - Length - 2.0 (1-3) - 2.2 (2-3) .052 - Meatus - 2.1 (0-3) - 2.9 (2-3) < .001 - Glans - 2.5 (2-3) - 2.6 (2-3) .0081 - Skin - 2.2 (1-3) - 2.3 (2-3) - Axis - 2.3 (1-3) - 2.6 (2-3) - Appearance - 2.4 (2-3) - 2.3 (2-3) Erectile function (IIEF) 31.4 (14-35) 33.7 (30-35) .04 Self-esteem (Rosenberg) 30.6 (24-37) 35.8 (28-40) .0001 Quality of life (Euroqol5D) 94.3 (80-100) 91.3 (65-100) .29 Table 2. Comparison of the scores of the patients and the control group. Abbreviations: PPPS: Pediatric Penile Perception Score; IIEF: International Index of Erectile Functiona The continuous variables were compared using a parametric test (Student’s t-test) and a non-parametric test (Mann-Whitney test) Long-term outcomes of hypospadias-Belgacem et al. Vol 18 No 5 September-October 2021 539 additional postoperative complications were noted, nor did any further surgery take place. None of the patients indicated that they suffered from urinary functional im- pairment. One patient died of an unrelated cause. Five patients returned to the dedicated consultation unit for a medical examination and a uroflowmetry test. Only 15 patients complied with the study requirements by returning the questionnaire after a median follow-up of 22 years (IQR 20,26), their median age was 24.3 years (IQR 20,27). According to surgeons, the median short-term postoperative PPS score was 17.8 (range 17- 18), which was representative of the MAGPI population 17.7 (range 17-18) in this study period. Their outcomes were compared to those for a population of 15 matched circumcised men (median age 28.8 years (IQR 21,38)) and 15 matched uncircumcised men (median age 27.2 years (IQR 21,39)). No differences were found for the scores of the four parameters that were assessed between the circumcised and the uncircumcised population (Table 1). Thus, we decided to compare the operated patients with all of the controls by combining both the circumcised and the un- circumcised men. The control subjects were older than the patients (28.0 vs. 24.3 years of age; p < .05), although the median age at the time of the surgery was the same for both groups (Table 2). There was not a significant difference be- tween the patients and the control group in terms of the quality of life (p = .29). There was a significant differ- ence, however, regarding the penile cosmetic self-per- ception (13.3 vs. 15.8; p < .01). This difference was mainly due to the following items: the length (2.0 vs. 2.2; p = .052), the meatus (2.1 vs. 2.9; p < .001), and the axis (2.3 vs. 2.6; p = .0081) of the penis (Table 2). There was also a significant difference in the self-es- teem score (30.6 vs. 35.8; p < .01) and the erectile func- tion score (31.4 vs. 33.7; p = .04). Two patients had not yet had a sexual partner. Lower self-esteem correlated with poor genital self-perception (r = .92; p < .01). No association could be found between lower self-esteem and reduced erectile function. DISCUSSION Our results indicate that adult men operated on for dis- tal hypospadias have lower self-esteem and penile per- ception scores compared with controls. The quality of life was comparable to that of the controls, as has also been reported for a population of patients with heter- ogeneous operated hypospadias.(4) This study was the first to present long-term self-perception outcomes. The authors pointed out that the limitations of their study comprised the late age at which the surgical procedure was performed (more than 4 years of age), heterogene- ous types of hypospadias and surgical techniques, and the exclusive use of circumcised men as controls. There is a paucity of studies regarding long-term fol- low-up of hypospadias because the transition between pediatric and adult urology is not systematic for most patients.(15-17) The most common reason for genital dis- satisfaction in hypospadias patients is inadequate penile size, while the experience with masturbation appears to be similar to that of the general population.(18) It is well known that men with corrected hypospadias suffer from sexual inhibition and fear of being ridiculed, while those with proximal hypospadias are also more likely to be dissatisfied with their genital appearance.(17,19-21) No long-term-follow studies are available specifically for distal hypospadias. The present study showed that the perceived quality of life was the same for the patients, despite reduced erectile function compared to the con- trols. The main bias in regard to the erectile function item was that two of the patients had not yet engaged in sexual intercourse. There was no difference, however, in the overall erectile function score when these two pa- tients were excluded from the patient group. Recent studies have found that laypersons are not trou- bled by the penile appearance after hypospadias repair and that women perceive the genitals of men with distal hypospadias as being similar to the genitals of non-af- fected, circumcised men.(22,23) Similarly, men with non-operated hypospadias often find that some of their sexual partners or medical professionals are not aware of their hypospadias. In the present study, the patients expressed clear displeasure regarding the length, mea- tus, and axis of their penis, whereas the overall appear- ance was rated as being similar in both groups.(24,25) As described previously, these concerns did not appear to impact the overall quality of life.(4) Particularly in case of distal hypospadias, it is likely that the main reason for the stated low level of satisfaction with their penile appearance could be considered to be part of their lower overall self-esteem. Self-esteem was distinctly lower in the patients: 8 (53%) of them had a score below 30, which means that specific psycholog- ical support is required. The underlying reason for this is not clear, however. Indeed, the role of surgical treat- ment may be of little or no relevance as circumcised patients do not exhibit the same psychological profile. Most of the patients contacted by phone stated that they did not remember undergoing the surgery during their childhood. However, the emotional reaction of the par- ents in regard to the hypospadias appears to be critically important for the patients’ psychological development. (26) This information may be of considerable relevance to practitioners who treat patients with hypospadias and their families with the aim of preventing the develop- ment of feelings of shame and negative genital percep- tion.(27,28) When these patients undergo counseling, they should be provided support to develop a positive genital self-perception, as poor genital self-perception has been shown to correlate with an impaired mental health-re- lated quality of life.(4) Our data confirm the need for long-term follow-up to allow patients to address con- cerns regarding their genitals.(2,6) The main limitations of this study are the small number of patients and the possibility of selection bias for pa- tients who agreed to participate in the survey. Another limitation is the absence of an ideal control group of adult patients with hypospadias but who did not under- go surgical correction. CONCLUSIONS The findings of the current study confirm poor genital self-perception of adult patients operated from distal hypospadias. This poor genital perception correlated with lower self-esteem. Consequently, for clinical man- agement, we suggest that adult hypospadias patients are made more aware of the fact that their penile ap- pearance is often a non-issue for laypersons. Early and long-term follow-up of patients and their parents could prevent this negative genital and overall self-percep- tion from taking hold. The main limitation of this study Long-term outcomes of hypospadias-Belgacem et al. Pediatric Urology 540 relates to its long follow-up retrospective design with many consecutive biases. 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