The repair of proximal hypospadias remains a challenge for pediatric urologists. Current controversial topics center around the use of 1- or multiple stage techniques, methods of correction of the penile curvature and preservation or not of the urethral plate among others. The aim of this Research topic is to attract articles from experts in the field to paint a picture of the current trends in this area.

Kinder- und Jugendkrankenhaus AUF DER BULT

University of Miami

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Frontiers in Pediatrics

Pediatric Urology

Frontiers in Surgery

Surgical Management Options in Proximal Hypospadias

This research topic intended to highlight different philosophies and techniques for the correction of proximal hypospadias, a topic that continues to generate great controversy among pediatric urologists. Unfortunately, there were only four papers accepted to be included in this collection.One paper dealt with the results of one-stage repair of mid- and proximal hypospadias, a second paper dealt with a technique to facilitate second-stage repairs when the urethral plate created in the first stage with a free graft was difficult to tabularize, a third paper reviewed the current status of tissue engineering for urethral reconstruction, and the fourth one dealt with the results of a modified Mathieu technique for repair of the megameatus/intact prepuce variant of hypospadias.Authors from Germany (Hannover and Berlin) reported the results (measured by reoperation rate) in 49 children operated with a technique first described by the senior author in 1996 and followed for 2 years after the operation (<a href="https://doi.org/10.3389/fped.2018.00019">González et al.</a>). The technique consists of the preservation of the urethral plate whenever possible (48 of the 49 patients) and then creating an onlay urethroplasty using the outer preputial layer of a total preputial flap transposed ventrally through a buttonhole in its pedicle to avoid rotation. The penile curvature was corrected with chordectomy alone in 10 patients; 38 patients required a dorsal plication of the tunica albuginea and 1 patient required an additional ventral dermal graft. Success, defined as a straight penis, a glanular meatus, and the absence of voiding symptoms, was achieved in 77.5% of the patients with a single procedure. Complications included urethrocutaneous fistulas in 16% of the patients, glans dehiscence in 6% of the patients, and recurrent chordee in one patient. Of the 13 patients that had uroflow examinations, 11 had bell-shaped curves. This report would suggest that one-stage repairs can achieve good results with fewer operations than the more commonly used staged techniques. Shortcomings of this report include relative short follow-up and lack of objective cosmetic evaluation.A group from Vienna reported on 35 consecutive patients from a cohort of 250 hypospadias cases who completed both steps of a staged repair (<a href="https://doi.org/10.3389/fped.2019.00060">Tonnhofer et al.</a>). The original idea in this report, however, is based on the fact that in two-thirds of the patients, a midline incision of the graft was considered necessary to achieve tubularization during the second stage. With a mean follow-up of 18 months, the complication rate was 22%, similar to that reported in the previously commented paper for a single-stage repair. The complication rate in this series was greater with the use of buccal mucosa than with the use of a free preputial graft. Complication rate in cases in which the plate was not incised (presumably because it was wider or of better “quality”) was just 8.3%. Despite these results, the authors conclude that a two-stage repair is the procedure of choice in the correction of severe hypospadias. Other shortcomings of this report include short follow-up, lack of objective criteria to decide to incise the plate, and lack of objective evaluation of the results of the urethroplasty, such as uroflow studies or calibration. Therefore, this paper adds a trick to facilitate the second stage, but the authors fail to demonstrate the real value of the maneuver. Perhaps it would be more productive to emphasize the need to have a very generous graft in the first stage and to wait until the graft is soft and pliable to perform the second stage.A surgeon from Boston describes his experience with the use of a metal-based flap to repair the megameatus intact prepuce variant of hypospadias (MIP) (<a href="https://doi.org/10.3389/fped.2018.00055">Cendron</a>). Twenty-five patients operated in a 10-year period were divided into two groups. The first 10 cases underwent simple tubularization of the plate. One fistula developed. The next 15 cases underwent repair using a metal-based flap. There were no fistulas, but one patient experienced dehiscence of the glans. The author favors the metal-based flap technique, because in this series, it eliminated injury of the urethral plate during the dissection. Of course, the numbers are too small to draw definitive conclusions, but given the rarity of this condition, the author's opinion is worth considering.A paper by authors from Qatar and Denmark analyzed the current knowledge of tissue-engineered material for urethral replacement (<a href="https://doi.org/10.3389/fped.2017.00283">Abbas et al.</a>). The clinical experience cited is limited, but tissue engineering will certainly become a reality in the future that will benefit patients with undesirable results from surgical misadventures where local tissues are not available for reconstruction. The authors conclude that despite many significant advances, the search for a suitable tissue engineering approach for use in routine clinical applications continues.A vital part of successful correction of severe hypospadias is the straightening of the penis. Although not included in this research topic, a recent article in Frontiers provides a comprehensive review of this topic and is highly recommended for surgeons interested in this topic (<a href="#B1">1</a>).All authors listed have made a substantial, direct and intellectual contribution to the work, and approved it for publication.The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.

Editorial: Surgical Management Options in Proximal Hypospadias

Purpose: In severe hypospadias staged repair is commonly used and it is regarded as feasible, safe, and durable. In this article we want to describe the results of a modification of the staged repair: a midline incision of the graft during the second stage.Materials and Methods: This is a consecutive single team (2 surgeons) retrospective series. Between 2014 and 2017, 250 patients underwent hypospadias repair, among them 35 patients that had primary staged hypospadias surgery with completed first and second stage repair. 24 (68.6%) cases received a preputial skin graft and 11 (31.4%) buccal mucosa graft. Median age at first stage was 1.5 (0.5–22.1) years, mean time between first and second stage operation was 0.72 (0.4–1.76) years. Follow up rate was 100%, mean follow up period was 1.50 (0.4–3.8) years.Results: The total complication rate was 22.9%. In buccal mucosa repair the complication rate was 36.4% and in preputial graft repair the complication rate was 16.7%, respectively. In 23 patients (65.7%) during second stage urethroplasty a midline incision was performed (8 glandular graft, 15 penile graft, 6 at level of urethral opening). Complication rate in non-incised urethroplasty was 8.3%, in incision at glandular level 37.5%, in incision at penile level 13.3% and in incision at urethral opening 16.7%, respectively.Conclusions: Two stage repair is the method of choice in the correction of severe hypospadias. In selected cases a midline incision of the graft is feasible and can be applied if needed. Randomized studies will be needed to evaluate the true benefit of incising the graft.

Purpose: In severe hypospadias staged repair is commonly used and it is regarded as feasible, safe, and durable. In this article we want to describe the results of a modification of the staged repair: a midline incision of the graft during the second stage.Materials and Methods: This is a consecutive single team (2 surgeons) retrospective series. Between 2014 and 2017, 250 patients underwent hypospadias repair, among them 35 patients that had primary staged hypospadias surgery with completed first and second stage repair. 24 (68.6%) cases received a preputial skin graft and 11 (31.4%) buccal mucosa graft. Median age at first stage was 1.5 (0.5–22.1) years, mean time between first and second stage operation was 0.72 (0.4–1.76) years. Follow up rate was 100%, mean follow up period was 1.50 (0.4–3.8) years.Results: The total complication rate was 22.9%. In buccal mucosa repair the complication rate was 36.4% and in preputial graft repair the complication rate was 16.7%, respectively. In 23 patients (65.7%) during second stage urethroplasty a midline incision was performed (8 glandular graft, 15 penile graft, 6 at level of urethral opening). Complication rate in non-incised urethroplasty was 8.3%, in incision at glandular level 37.5%, in incision at penile level 13.3% and in incision at urethral opening 16.7%, respectively.Conclusions: Two stage repair is the method of choice in the correction of severe hypospadias. In selected cases a midline incision of the graft is feasible and can be applied if needed. Randomized studies will be needed to evaluate the true benefit of incising the graft.

Midline Incision of a Graft in Staged Hypospadias Repair–Feasible and Durable?

IntroductionThe megameatus intact prepuce (MIP) variant of hypospadias is a rare variant of hypospadias that is diagnosed either early at the time of circumcision or later as the foreskin is retracted. The true incidence of the anomaly is difficult to determine precisely as some patient never come to medical attention but is felt to under 5% of all cases of hypospadias. The purposes of this study are to review the embryology and clinical findings of MIP and then, in light of a personal experience, present a series of patients evaluated for MIP who were treated with a modification of the Mathieu technique.</sec>Materials and methodsA PubMed search of all articles in the MIP variant of hypospadias was carried out followed by an exhaustive review of the literature. The charts of all patients evaluated and treated at Boston Children’s Hospital by MC between 2007 and 2017 were reviewed retrospectively. The patients were divided into two groups: those who underwent the standard procedure and those who underwent a repair using a modification of the Mathieu procedure using an inframeatal flap.</sec>ResultsThe embryologic explanation of the MIP variant is not clear but failure of the distal, glanular portion of the urethra to tubularize results in spectrum of abnormality characterized by a deep glanular groove and an abnormal opening of the urethra anywhere from the mid-glans to a subcoronal location. Surgical repair is complicated by a wide distal urethra which may be injured if not properly identified. Overall good outcomes were noted with one patient experiencing a urethra cutaneous fistula in the first group and one patient having a mild glans dehiscence in the second.</sec>ConclusionThe MIP variant of hypospadias is a rare variant of hypospadias that presents as a spectrum of urethral anomaly. Surgical repair may not always be necessary but if surgical repair is carried out, the Mathieu technique modification may offer better anatomic delineation of the urethra and will provide an extra layer of tissue to cover the reconstructed urethra. Low complication rates should be expected with adequate functional outcome such as a normal urinary stream. In addition, criteria for selecting patients for surgical repair are provided.</sec>

Introduction: The megameatus intact prepuce (MIP) variant of hypospadias is a rare variant of hypospadias that is diagnosed either early at the time of circumcision or later as the foreskin is retracted. The true incidence of the anomaly is difficult to determine precisely as some patient never come to medical attention but is felt to under 5% of all cases of hypospadias. The purposes of this study are to review the embryology and clinical findings of MIP and then, in light of a personal experience, present a series of patients evaluated for MIP who were treated with a modification of the Mathieu technique.Materials and methods: A PubMed search of all articles in the MIP variant of hypospadias was carried out followed by an exhaustive review of the literature. The charts of all patients evaluated and treated at Boston Children’s Hospital by MC between 2007 and 2017 were reviewed retrospectively. The patients were divided into two groups: those who underwent the standard procedure and those who underwent a repair using a modification of the Mathieu procedure using an inframeatal flap.Results: The embryologic explanation of the MIP variant is not clear but failure of the distal, glanular portion of the urethra to tubularize results in spectrum of abnormality characterized by a deep glanular groove and an abnormal opening of the urethra anywhere from the mid-glans to a subcoronal location. Surgical repair is complicated by a wide distal urethra which may be injured if not properly identified. Overall good outcomes were noted with one patient experiencing a urethra cutaneous fistula in the first group and one patient having a mild glans dehiscence in the second.Conclusion: The MIP variant of hypospadias is a rare variant of hypospadias that presents as a spectrum of urethral anomaly. Surgical repair may not always be necessary but if surgical repair is carried out, the Mathieu technique modification may offer better anatomic delineation of the urethra and will provide an extra layer of tissue to cover the reconstructed urethra. Low complication rates should be expected with adequate functional outcome such as a normal urinary stream. In addition, criteria for selecting patients for surgical repair are provided.

The Megameatus, Intact Prepuce Variant of Hypospadias: Use of the Inframeatal Vascularized Flap for Surgical Correction

AimsTo report current results of preputial flap onlay urethroplasty using the principle of the total preputial flap (TPF) for the one-stage repair of mid- and proximal hypospadias.</sec>MethodsThis study was a retrospective chart review of patients in a prospectively kept database of all hypospadias operations performed at two institutions from January 1 2011 to August 2017. Inclusion criteria: all patients who underwent hypospadias repair using a preputial only flap urethroplasty based on the principle of the TPF. Demographic data, duration of follow-up, complications, and reoperations were recorded. A successful result was considered to be a straight penis, a glanular meatus, and absence of voiding symptoms. Whenever possible an uroflow was obtained during the follow-up visits.</sec>ResultsForty-nine children met the inclusion criteria. All patients had marked penile curvature. Three patients had chromosomal abnormalities. The mean age at the time of surgery was 22 months (11–110) and the mean duration of follow-up 23.4 months (1–79). In 48 cases, the urethral plate could be preserved without dividing it. The penile curvature was corrected with chordectomy alone in 10 patients, 38 required a dorsal plication of the tunica albuginea, and 1 required an additional ventral dermal graft. In 38 patients (77.5%), the initial operation was successful, and no further operations were needed. There were eight urethrocutaneous fistulas, three dehiscences of the glans approximation. One patient suffered a wound infection and partial loss of the flap.</sec>ConclusionOne-stage repair of mid- and proximal hypospadias preserving the urethral plate and using a TPF for the urethroplasty and coverage of the ventral penis is successful in 77.5% of cases. Complications in the remaining patients were easily managed or did not require treatment. Compared to a planned two-stage approach, the technique described in this report resulted in significantly fewer procedures till complete resolution of the problem.</sec>

Aims: To report current results of preputial flap onlay urethroplasty using the principle of the total preputial flap (TPF) for the one-stage repair of mid- and proximal hypospadias.Methods: This study was a retrospective chart review of patients in a prospectively kept database of all hypospadias operations performed at two institutions from January 1 2011 to August 2017. Inclusion criteria: all patients who underwent hypospadias repair using a preputial only flap urethroplasty based on the principle of the TPF. Demographic data, duration of follow-up, complications, and reoperations were recorded. A successful result was considered to be a straight penis, a glanular meatus, and absence of voiding symptoms. Whenever possible an uroflow was obtained during the follow-up visits.Results: Forty-nine children met the inclusion criteria. All patients had marked penile curvature. Three patients had chromosomal abnormalities. The mean age at the time of surgery was 22 months (11–110) and the mean duration of follow-up 23.4 months (1–79). In 48 cases, the urethral plate could be preserved without dividing it. The penile curvature was corrected with chordectomy alone in 10 patients, 38 required a dorsal plication of the tunica albuginea, and 1 required an additional ventral dermal graft. In 38 patients (77.5%), the initial operation was successful, and no further operations were needed. There were eight urethrocutaneous fistulas, three dehiscences of the glans approximation. One patient suffered a wound infection and partial loss of the flap.Conclusion: One-stage repair of mid- and proximal hypospadias preserving the urethral plate and using a TPF for the urethroplasty and coverage of the ventral penis is successful in 77.5% of cases. Complications in the remaining patients were easily managed or did not require treatment. Compared to a planned two-stage approach, the technique described in this report resulted in significantly fewer procedures till complete resolution of the problem.

Results of Onlay Preputial Flap Urethroplasty for the Single-Stage Repair of Mid- and Proximal Hypospadias

Hypospadias, characterized by misplacement of the urinary meatus in the lower side of the penis, is a frequent birth defect in male children. Because of the huge variation in the anatomic presentation of hypospadias, no single urethroplasty procedure is suitable for all situations. Hence, many surgical techniques have emerged to address the shortage of tissues required to bridge the gap in the urethra particularly in the severe forms of hypospadias. However, the rate of postoperative complications of currently available surgical procedures reaches up to one-fourth of the patients having severe hypospadias. Moreover, these urethroplasty techniques are technically demanding and require considerable surgical experience. These limitations have fueled the development of novel tissue engineering techniques that aim to simplify the surgical procedures and to reduce the rate of complications. Several types of biomaterials have been considered for urethral repair, including synthetic and natural polymers, which in some cases have been seeded with cells prior to implantation. These methods have been tested in preclinical and clinical studies, with variable degrees of success. This review describes the different urethral tissue engineering methodologies, with focus on the approaches used for the treatment of hypospadias. At present, despite many significant advances, the search for a suitable tissue engineering approach for use in routine clinical applications continues.

Hypospadias, characterized by misplacement of the urinary meatus in the lower side of the penis, is a frequent birth defect in male children. Because of the huge variation in the anatomic presentation of hypospadias, no single urethroplasty procedure is suitable for all situations. Hence, many surgical techniques have emerged to address the shortage of tissues required to bridge the gap in the urethra particularly in the severe forms of hypospadias. However, the rate of postoperative complications of currently available surgical procedures reaches up to one-fourth of the patients having severe hypospadias. Moreover, these urethroplasty techniques are technically demanding and require considerable surgical experience. These limitations have fueled the development of novel tissue engineering techniques that aim to simplify the surgical procedures and to reduce the rate of complications. Several types of biomaterials have been considered for urethral repair, including synthetic and natural polymers, which in some cases have been seeded with cells prior to implantation. These methods have been tested in preclinical and clinical studies, with variable degrees of success. This review describes the different urethral tissue engineering methodologies, with focus on the approaches used for the treatment of hypospadias. At present, despite many significant advances, the search for a suitable tissue engineering approach for use in routine clinical applications continues.