34th ESPU Congress in Naples, Italy

S15: HYPOSPADIAS 1

Moderators: Anne Françoise Spinoit (Belgium), Haytham Badawy (Egypt)

ESPU Meeting on Friday 19, April 2024, 09:00 - 10:05


09:00 - 09:03
S15-1 (OP)

PREVALENCE AND AGE AT DIAGNOSIS OF MALES WITH HYPOSPADIAS - A POPULATION-BASED NATIONWIDE EPIDEMIOLOGICAL STUDY

Tina LEUNBACH 1, Agnethe BERGLUND 2, Andreas ERNST 3, Gitte M. HVISTENDAHL 4, Yazan F. RAWASHDEH 5 and Claus GRAVHOLT 6
1) Aarhus University Hospital and Aarhus University, Department of Urology, Section of Paediatric Urology and Department of Clinical Medicine, Aarhus, DENMARK - 2) Aarhus University Hospital, Department of Molecular Medicine and Department of Clinical Genetics, Aarhus, DENMARK - 3) Aarhus University Hospital and Aarhus University, Department of Urology, Section of Paediatric Urology and Department of Public Health, Research Unit for Epidemiology, Aarhus, DENMARK - 4) Aarhus University Hospital, Department of Urology, Section of Pediatric Urology, Aarhus, DENMARK - 5) Aarhus University Hospital, Department of Urology, Section of Pediatric Urology,, Aarhus, DENMARK - 6) Aarhus University Hospital, Department of Endocrinology and Internal Medicine and Department of Molecular Medicine, Aarhus, DENMARK

PURPOSE

The presumed multifactorial etiology of hypospadias remains incompletely understood. Secular trends in disease patterns may elucidate associated risk factors. The reported prevalences of hypospadias present conflicting findings.

We aimed to assess the prevalence of hypospadias and its secular trend in Denmark.   

MATERIAL AND METHODS

Males with a hypospadias diagnosis in the Danish National Patient Registry (DNPR) initiated in 1977 were identified. Information from the birth cohorts in Denmark (1901-2019) was used to calculate the prevalence (number of males with hypospadias per 100,000 newborn boys). For males born after 1977 the first recording in the DNPR defined the diagnostic age. Temporal trends were analyzed by linear regression.

RESULTS

From 1977, n=9,187 males, born from 1901-2019, were recorded with hypospadias in the DNPR. The prevalence rose until 1977. From 1977-2006, the mean prevalence rose to 502.6 (95% CI 440.4-564.9, p<0.001) and peaked at 847.2 in 2007. From 2008-2018, the mean prevalence stabilized at 773.8 (95% CI 738.1-809.5, p=0.3231). The mean diagnostic age (1977-2019) was 1.9 years (95% CI 1.8-2.0) and showed a slight decreasing temporal trend (p<0.001).

CONCLUSIONS

This study reports a high prevalence of hypospadias currently around 800 per 100,000 newborn boys. Similar prevalences have been reported in Sweden and in some states in the USA. Multiple studies have reported lower prevalences. Methodological heterogeneity between studies challenges the reported differences across the world. Successful surveillance reflecting true estimates depends on consistent and effective ascertainment as is the case in Denmark.


09:03 - 09:06
S15-2 (OP)

RELATIONSHIP OF HYPOSPADIAS AND SOURCES OF POLLUTION IN A STATIC COMMUNITY IN NEW ENGLAND

Borivoj GOLIJANIN, Emily BARRY, Rachel GREENBERG, Anthony CALDAMONE and Hsi-Yang WU
The Minimally Invasive Urology Institute, The Miriam Hospital, Warren Alpert Medical School of Brown University, Urology, Providence, USA

PURPOSE

It is thought that endocrine disrupting environmental factors, which may increase risk for hypospadias, can be found industrial waste storage and processing facilities. This study investigates whether hypospadias in an industrial region in New England has a geographic distribution that is related to sources of pollution.

MATERIAL AND METHODS

All hypospadias cases at a single hospital system from 2015 to 2018 were retrospectively reviewed. These years were selected due to the availability of pollution records. Cases were categorized into type (proximal, midshaft, distal), and then geocoded and mapped. Regional waste storage and processing facilities were also mapped including. Geographic distribution of cases was compared to the distribution of the pollution sites. Correlation between type of hypospadias with proximity of pollution sites was evaluated.

RESULTS

Types of hypospadias (n=113) included 20.4% proximal, 10.6% midshaft , and 69% distal. There was no significant association of incidence of cases and pollution source. Stratifying by type of hypospadias, 48.4% of clusters were comprised of distal hypospadias cases only, they were found farther from the sites of pollution. 51.6% of clusters were comprised of proximal and midshaft cases only. Although non-significant, they were found in areas closer to sources of pollution, compared to distal cases.

CONCLUSIONS

Although distance to an environmental pollutant was not significant, there is a clear geographic separation of the type of hypospadias found in this region. Additional study of the causes of hypospadias, including amount of exposure, and genetic factors, is warranted to explain the geographic separation and etiology of this congenital anomaly.


09:06 - 09:09
S15-3 (OP)

★ UROLOGICAL CANCER RISK IN CHILDREN AND ADULTS BORN WITH HYPOSPADIAS USING A SWEDISH REGISTER-BASED COHORT

Lottie PHILLIPS 1, Cecilia LUNDHOLM 2, Catarina ALMQVIST 2, Anna SKARIN NORDENVALL 2 and Agneta NORDENSKJÖLD 1
1) Karolinska Institutet, Women's and Children's Health, Solna, SWEDEN - 2) Karolinska Institutet, Medical Epidemiology and Biostatistics, Stockholm, SWEDEN

PURPOSE

The risk of urological cancer in individuals with hypospadias could be increased due to different mechanisms including mutual aetiology, androgen dysfunction, and recurring inflammation or infection. We therefore aimed to investigate whether urological cancer risk is increased in children and adults born with hypospadias.

MATERIAL AND METHODS

We used national Swedish register data to study a population-based cohort of individuals born 1964-2018, as well as adults born from 1940. Outcomes were defined using diagnostic- and morphological codes for cancer subtypes. Associations were measured using Cox proportional-hazards regression analysis.

RESULTS

In total, we identified 4 284 902 individuals, including 17 549 with a diagnosis of hypospadias. In children, we found an association with Wilms' tumour which may relate to shared genetics. Throughout life, we found an increased risk of testicular cancer in boys and men with hypospadias (hazard ratio 2.04, 95% confidence interval 1.42-2.92). The risk increase was greater in those with proximal hypospadias and the association remained when only studying those without cryptorchidism. In adults, we further found an increased risk of bladder and urethral cancer, but no statistically significant association with prostate cancer. However, the absolute risk of cancer in individuals with hypospadias was low, which was reflected by the small number of men with both conditions.

CONCLUSIONS

Hypospadias, especially proximal hypospadias, is associated with an increased risk of testicular cancer that is not explained by a co-occurrence with cryptorchidism. Our results further indicate an increased risk of lower urinary tract cancers which merits further investigation. More research is needed to understand which specific subgroups of individuals may be at risk and why.


09:09 - 09:12
S15-4 (OP)

MECHANISTIC ASSESSMENT OF PENILE BLOOD FLOW FOLLOWING CAUDAL BLOCK

Adam ADLER 1, Vinaya BHATIA 2, Arvind CHANDRAKANTAN 1 and Paul AUSTIN 3
1) Texas Children's Hospital, Baylor College of Medicine, Department of Anesthesiology, Perioperative and Pain Medicine, Houston, USA - 2) University of Wisconsin, Urology, Madison, USA - 3) Texas Children's Hospital, Baylor College of Medicine, Urology, Houston, USA

PURPOSE

In adults, neuraxial anesthesia cause hypotension primarily due to blockade of sympathetic nerve fibers which result in peripheral vasodilation. These hemodynamic changes have not been seen in young children and we hypothesize that penile blood flow would not be altered from caudal block administration. Our objective is to perform a mechanistic assessment to characterize penile arterial and venous blood flow in association with caudal block given the past controversial association with hypospadias surgery.

MATERIAL AND METHODS

A prospective study was conducted in children undergoing penile surgery. The penile artery and venous flow velocity were assessed using Doppler ultrasound pre and post caudal block. Exclusion criteria included: patients with allergy to local anesthetic, patients with cardiovascular issues including the use of cardiac medications, contradictions to caudal block (sacral dimple or local rash or infection), inability to attain ultrasound velocity measurements due to anatomic limitations of the penile vasculature and parental refusal.

RESULTS

Ten healthy patients were included in the study with a mean age and weight of 1.3 years and 10.7 kg respectively. There was no significance difference in mean arterial blood flow velocity 0.18 m/s, (95% CI: -1.55, 1.92; p=0.81) or mean dorsal penile vein flow velocity 0.11 m/s, (95% CI: -1.12, 1.33; p=0.84) prior to and following caudal block in our pediatric cohort. There was no correlation in the direction of change between the arterial flow and venous flow from before and after caudal block (R2=0.03)

CONCLUSIONS

Despite previous controversy surrounding the use of caudal block in children undergoing hypospadias correction, we did not identify an association between penile arterial or venous blood flow and performance of caudal block in children undergoing penile surgery. Our study presents encouraging findings that no significant hemodynamic change appears to result from caudal block.


09:12 - 09:25
Discussion
 

09:25 - 09:28
S15-5 (OP)

WHAT IS THE INCIDENCE OF CHORDEE IN DISTAL HYPOSPADIAS?

Melissa MCGRATH 1, Yaqoub JAFAR 1, Ziyad ALZAHRANI 1, Bruno LESLIE 1 and Luis H. BRAGA 2
1) McMaster University, Surgery-Urology, Hamilton, CANADA - 2) McMaster University - McMaster Children's Hospital, Department of Surgery / Urology, Hamilton, CANADA

PURPOSE

The implications of chordee measurement in distal cases and its impact on the choice of surgical technique and outcomes have yet to be objectively studied. We aimed to determine the rate of chordee in distal hypospadias and how its severity affected the decision-making to proceed with single vs staged repair.

MATERIAL AND METHODS

Prospective study(2016-2022) of consecutive boys with distal hypospadias based on the initial meatal location. All patients had erection test to measure the degree of chordee using a photograph angle app. We captured age at surgery, meatal location, glans groove depth, degree of chordee before and after degloving, midline raphe deviation(MRD) and ventral skin deficiency(VSD).

RESULTS

Of 318 cases, 237 had distal penile, 58 coronal, and 23 glanular hypospadias. Chordee was identified in 68% (216/318) patients, 12% had flat/no glans groove and 25% VSD. Before degloving, chordee was <30 in 78%, 30-70 in 20%, >70 in 2%. After degloving, 75% had no chordee, 19% had<30, and 6% had>30. Of those with chordee>30 after degloving (mean=368.6), all had flat/no glans groove, 10(71%) had MRD, 4(29%) VSD and 6(43%) proximal division of spongiosum. 75% of them underwent a dorsal inlay graft(DIG) or staged repair.

CONCLUSIONS

A minority(6%) of distal hypospadias patients have chordee >30(mean=36) after degloving. 70% of them had flat/no glans groove and MRD, which is suggestive of a more severe(proximal) phenotype, calling for urethral plate augmentation(DIG or staged repair). Be aware of these "distal" hypospadias with hidden proximal characteristics, as failure to identify these phenotypes may result in inadequate chordee correction and worse outcomes.


09:28 - 09:31
S15-6 (OP)

COMPARISON OF HYPOSPADIAS PHENOTYPE PIXEL SEGMENTATION TO GMS SCORE

Nicolas FERNANDEZ 1, Joshua CALDWELL 2, Meghan NOONAVATH 2, Margarett SHNORHAVORIAN 2, Adam MAXWELL 2 and Viktoriya SAPKALOVA 2
1) Seattle Childrens Hospital - University, Urology, Seattle, USA - 2) Seattle Childrens Hospital - University, Seattle, USA

PURPOSE

Introduction:Glans-Urethral Meatus-Shaft (GMS) score has been adopted to standardize hypospadias classification. While extremely subjective, GMS has been widely used to classify the severity of the phenotype to predict surgical outcomes. The use of digital image analysis has proven to be feasible. Nonetheless, the creation of these image recognition algorithms is subjective. To reduce a subjective input, we propose a novel approach using digital image pixel analysis comparing it the GMS score. Our hypothesis is that pixel cluster segmentation can discriminate between favorable and unfavorable anatomy.

MATERIAL AND METHODS

Methods:A total of 148 patients with different types of hypospadias were classified independently by experts following the GMS score into “favorable”, “moderately favorable” and “unfavorable”. From there, 592 images were generated using digital image segmentation. 584 were included for final analysis. For each image, the region of interest was segmented into “glans,” “urethral plate,” “foreskin” and “periurethral plate”.  Values obtained for each segmented region using statistical pixel k-means cluster analysis were compared to the GMS score using an ANOVA analysis.

RESULTS

Results:Analysis of image segmentation demonstrated that k-means pixel cluster analysis discriminated “favorable” vs “unfavorable” urethral plates. There was a significant difference between scores when comparing the GG and GM groups (p = 0.03) and GG and GP groups (p = 0.05). Pixel cluster analysis could not discriminate between “moderately favorable” and “unfavorable” urethral plates.       

CONCLUSIONS

Conclusions:We found significant pairwise difference between favorable and unfavorable anatomy. Digital image segmentation and statistical k-means cluster analysis can discriminate anatomical features following GMS score principles.


09:31 - 09:34
S15-7 (OP)

PLATE OBJECTIVE SCORING TOOL (POST); CORRELATION WITH POST-HYPOSPADIAS REPAIR COMPLICATIONS

Tariq Osman ABBAS 1, Ibrahim ADNAN 2, Jassim HATEM 3, Andrey BOYKO 4 and Sergei ZORKIN 5
1) Sidra Medicine, Urology, Doha, QATAR - 2) Hamad Medical Corporation,, Urology Department, Doha, QATAR - 3) Hamad Medical Corporation, Urology Department,, Doha, QATAR - 4) Pediatric Municipal Clinical Hospital,, Urology, Barnaul, RUSSIAN FEDERATION - 5) National Medical Research Center for Children's Health,, Urolohy, Moscow, RUSSIAN FEDERATION

PURPOSE

The Plate Objective Scoring System (POST) has been shown to reflect the configuration of urethral plate quality in distal hypospadias, serving as an assessment guide. Here we aim to appraise its correlation with post-distal hypospadias repair complications.

MATERIAL AND METHODS

Data were prospectively obtained from prepubertal boys who underwent primary hypospadias repair between January 2020 and February 2023. Both POST and GMS scores were measured by three different reviewers in triplicate and correlation with post-distal hypospadias repair complications was evaluated.

RESULTS

The POST ratios were strongly correlated with post- hypospadias repair complications in 121 patients. POST score values mean was 1.10 (range 0.5-1.62). The average total GMS score was 5.29 ± 1.36, and the medians of G, M, and S scores were 2, 2, and 1, respectively. Bivariate correlation analysis showed that the POST score was strongly accurate in predicting the chances of having complications, with a Pearson correlation coefficient of r = 0.821 (0.724-0.918) 95% CI. The cutoff value of POST with the highest specificity for the presence of post-operative complications was 1.2 and complications' incidence was 4.4% (2/45) in POST ≥ 1.2 and 25 % (19/76) in POST <1.2.

CONCLUSIONS

This study highlights the significance of the POST proportion as a surrogate sign for urethral plate quality for objectivity and accuracy in urethral plate evaluation, which in turn serves as an independent factor impacting outcomes in distal hypospadias repair. We demonstrated a statistically significant increase in the likelihood of any postoperative complication with a decrease in POST score.


09:34 - 09:45
Discussion
 

09:45 - 09:48
S15-8 (OP)

IMPROVING THE NURSING CONTRIBUTION TO HYPOSPADIAS SURGERY

Ellen BULLMAN 1, Hazem MOSA 2, Massimo GARRIBOLI 1, Anu PAUL 1, Arash TAGHIZADEH 1 and Pankaj MISHRA 1
1) Evelina children's Hospital London, Paediatric urology, London, UNITED KINGDOM - 2) Jenny Lind Children's Hospital, Paediatric urology, Norwich, UNITED KINGDOM

PURPOSE

Hypospadias is a common penile congenital anomaly that affects 1:200 boys. Hypospadias surgery has both short- and long-term complications. A significant nursing contribution is often required in the first post operative week. We aimed to improve the nursing knowledge and confidence about providing care to patients who recently underwent hypospadias surgery.

MATERIAL AND METHODS

A 7 question Questionnaire identifying staff nurses experience and knowledge of hypospadias nursing using a 5-point Likert scale. A structured nursing-centred workshop with emphasis on two clinical scenarios: hypospadias dressing removal and receiving a phone call from a parent of a hypospadias patient asking for advice. Training was delivered to band 5 (newly qualified) nurses on the nephro-urology ward of a tertiary paediatric urology centre over the period of 2 weeks.

RESULTS

15 nurses participated in the hypospadias training workshop. Questionnaire responses pre and post training were collected. Statistical analysis was performed using an independent t-test to compare responses pre and post training. A significant improvement in responses to the 7 questions was noted (table 1). An instructional video demonstration of hypospadias dressing removal by a paediatric urology clinical nurse specialist was produced to ensure continuity of the quality improvement initiative. A flow chart of the dressing removal procedure was also produced.

Pre-training

Post-training

P value

Question theme

Question 1

1.5

4.5

0.00

Types of hypospadias

Question 2

1.5

4.4

0.00

Hypospadias operations

Question 3

2.8

3.9

0.01

Dressing removal

Question 4

2.1

4.2

0.00

Short-term complications

Question 5

2.3

4.2

0.00

Managing postoperative complications

Question 6

2.3

4.2

0.00

Giving phone advice

Question 7

2.2

4

0.00

Hypospadias referral for escalation

Free text response

0.1

0.8

0.00

CONCLUSIONS

A focused nursing-centred training can increase nursing staff knowledge and confidence about managing patients with hypospadias. A sustainable mode of training delivery is needed to ensure training of newly qualified nurses.


09:48 - 09:51
S15-9 (OP)

A SIMPLE NOVAL TRICK TO PREVENT THE DRESSING FROM STOOL CONTAMINATION IN CHILDREN UNDERGO HYPOSPADIAS SURGERY: HYPOSPADIAS PAD

Abdurrahman ONEN
Dicle University Medical Faculty, Pediatric Surgery and Urology, Sur, TURKEY

PURPOSE

The clinical indication of a specific dressing is based upon the protective function and mechanical barrier of the tissues against contamination and reduction of infection in children underwent genital surgery. The correct choice of dressing is still challenging. The most important point is to prevent contamination of the surgical area with stool. We developed a special noval pad to prevent stool contamination in infant under go hypospadias repair.

MATERIAL AND METHODS

It is specially designed for infants who has not toilette training and wear diaper. The size of the this noval pad is 15 x 10 cm and has two adhesive wings each 10 cm. One adhesive wing is attached to perineum and distal part of scrotum, while the opposit wing is attached to the diaper. The adhesive long side of the pad is attached to each leg. We have educated and participated parent regarding this pad. Indications of our hypospadias pad is all kind of genital surgeries include hypospadias, epispadias, bladder exstrophy, burried penis, circumcision etc.

RESULTS

We prospectively used this special genital pad in 221 infants; 124 hypospadias, 13 epispadias, 16 burried penis, 5 bladder exstrophy and 63 circumsicion. We compared this group with 214 control infants who we did not use this pad that underwent a genital surgery. Stool contamination was observed in 4/221(1.8%) special pad group while it observed in 37/214(17.3%) control group.

CONCLUSIONS

The proposed material (hypospadias pad) seems to be a highly satisfactory alternative in improving post-operative care after hypospadias surgery. Its use is adequate for prevention of stool contamination after all kind of genital surgeries.


09:51 - 09:55
S15-10 (LOP)

★ ROS SCAVENGERS MAY IMPROVE GENITAL SKIN HEALING IN BOYS WITH HYPOSPADIAS

Angela LUCAS-HERALD 1, Samra HUSSAIN 1, Kirsty MCGINLEY 1, Rheure ALVES-LOPES 1, S Basith AMJAD 2, Martyn FLETT 3, Boma LEE 3, Mairi STEVEN 3, Stuart O'TOOLE 3 and S Faisal AHMED 1
1) University of Glasgow, Developmental Endocrinology Research Group, Glasgow, UNITED KINGDOM - 2) Royal Hospital for Children, Department of Paediatric Surgery, Glasgow, UNITED KINGDOM - 3) Royal Hospital for Children, Department of Paediatric Urology, Glasgow, UNITED KINGDOM

PURPOSE

Surgical repair of hypospadias is associated with high rates of surgical complications, including wound dehiscence. Boys with hypospadias have previously been demonstrated to have increased reactive oxygen species (ROS) compared to healthy controls. Our aim was to identify if cell migration and proliferation in genital skin is altered in boys with hypospadias, and whether this can be altered by antioxidants.

MATERIAL AND METHODS

Genital skin (GS) samples were collected from boys undergoing hypospadias repair (cases) or routine circumcision (controls). GS fibroblasts were grown until 80% confluence. Cells were imaged using an EVOS XL Core microscopeimmediately after a wound was made and 48 hours later, in the presence/absence of the ROS scavenger, Tempol. Cell migration was determined using ImageJ software. Cell proliferation was measured using a commercial Cell Count Kit-8 (Abcam, UK). 

RESULTS

Twenty four cases (median age (range) 1.8 (1.2, 6.3) years) and 28 controls (median age 1.6 (1.2, 6.1) years) were recruited. Location of hypospadias were distal in 14 (58%), proximal in 9 (38%) and midshaft in 1 (4%). Boys with hypospadias had impaired cell migration with reduced % wound closure at 48 hours (2.0 fold, p<0.0001) and reduced cell proliferation (1.3 fold, p=0.01). External Masculinisation Score was positively correlated with % wound closure (r=0.5, p<0.0001) and cell proliferation (r=0.3, p=0.002). Exposure to Tempol improved wound closure (1.5 fold, p=0.02) and cell proliferation (1.5 fold, p=0.02).

CONCLUSIONS

There is an association between wound healing and virilisation of the external genitalia in boys.  ROS scavengers improve cell migration and proliferation in boys with hypospadias.


09:55 - 10:05
Discussion