Foreskin Reconstruction or Conventional Circumcision in Hypospadias: A Systematic Review and Meta-analysis

ABSTRACT

Hypospadias is a common congenital anomaly of the male genitalia requiring surgical correction. During hypospadias repair, there are two broad approaches to deal with prepuce-foreskin reconstruction (FR) and conventional circumcision (CC). While FR preserves and reshapes the dorsal prepuce to mimic the normal appearance, CC involves excision or rearrangement of the prepuce leading to a circumcised appearance. The choice between these techniques is influenced by cultural, religious, and personal preferences; the techniques themselves differ in surgical complexity and complication risks. This systematic review and meta-analysis compares FR and CC in hypospadias repair focussing on surgical outcomes and complications. A comprehensive search of relevant randomized controlled trials and prospective studies were performed across PubMed, Embase, Scopus, and Cochrane Library. Studies were assessed for risk of bias using ROB-2 and ROBINS-I tools. Primary outcomes included urethra-cutaneous fistula (UCF), meatal stenosis (MS), and glans dehiscence (GD). Secondary outcomes included preputial complications and operative time. Both FR and CC yield comparable results in terms of major complications such as UCF, MS, and GD. Operative time shows variability, with no clear difference between the groups. However, FR carries a higher risk of preputial complications.

INTRODUCTION

Hypospadias is a common congenital anomaly of the male genitalia characterized by an abnormally positioned urethral meatus, ventral penile curvature (chordee), and a dorsally hooded foreskin. Its incidence in India is approximately 5.4 per 10000 live births.[1] The condition varies in severity, ranging from mild distal (glanular) to severe, proximal forms (scrotal, perineal). The associated functional and cosmetic concerns require surgical correction that aims at creating a straight phallus and a urinary stream from the tip of the glans.

Foreskin reconstruction (FR) and conventional circumcision (CC) are two common surgical approaches to manage the hooded foreskin during hypospadias repair. CC involves excision or rearrangement of the preputial skin that leaves the glans exposed much like after a circumcision; often, the foreskin is used in the surgical technique for hypospadias repair. FR involves preservation and reshaping of the dorsal prepuce to provide preputial covering to the glans after hypospadias repair akin to the normal appearance. FR is usually preferred for cultural, religious, or personal reasons.[2] Many parents and children consider the circumcised appearance as “unnatural.” In some traditions, circumcision is a part of a religious ritual; in them, FR may not be acceptable. Furthermore, some parents are happy with a straight phallus and passage of urine from the tip without any preference for FR or CC. CC may be associated with fewer complications related to FR like edema or ischemia of preserved prepuce.[3] The management of the foreskin in hypospadias surgery may sometimes play a crucial role in the psychosocial outcomes of patients. However, FR is technically complex, may require more time as compared to CC and not all surgeons are well versed in it. FR may have a higher risk of preputial complications compared to circumcision.[4] In fact, some patients of FR may require circumcision later for phimosis.[5] Although both methods are widely used, literature lacks high-quality evidence comparing the two. A systematic review and meta-analysis comparing the two approaches would provide evidence-based guidance for clinicians and families, and facilitate optimal surgical outcomes while respecting individual preferences. This analysis aims to compile such a body of evidence regarding FR and CC in hypospadias and offer guidance on the optimal surgical strategy.

Objectives

To systematically review and analyze the existing literature and data on FR and circumcision in hypospadias repair in children.

METHODS

Eligibility criteria

Randomized controlled trials (RCTs) and prospective studies comparing FR and circumcision techniques were included in the analysis. Retrospective comparative analysis, prospective comparative studies that observed exclusively long-term complications, case series, and case reports were excluded.

Population

Children who have undergone hypospadias repair. Intervention: Hypospadias repair with FR Comparison: Conventional hypospadias repair involving preputial excision or rearrangement to provide a circumcised appearance. Outcomes: Primary outcomes: incidence of urethro-cutaneous fistula (UCF), meatal stenosis (MS), and glans dehiscence (GD) postoperatively. Secondary outcomes: incidence of complications such as phimosis, wound dehiscence; and operative time.

Information sources

A thorough literature search was carried out across databases, including PubMed, Embase, Scopus, and the Cochrane Library. We also searched other sources like reference lists of included studies, clinical trial registries (e.g., ClinicalTrials.gov), and Gray literature sources such as conference abstracts.

Search strategy

We searched using a Boolean Search strategy-(Prepuce-preserving OR Foreskin Reconstruction OR Preputioplasty) AND (Circumcision) AND (Hypospadias).

Study selection

Two reviewers independently screened study titles and abstracts. Further, retrieved full-text articles were evaluated for eligibility, and any disagreements for the inclusion of any study were resolved by a third reviewer.

Data extraction

Two reviewers independently extracted the data from eligible studies using a standardized data collection form that included the following details like study characteristics – author, year, country, study design, sample size, patient demographics, intervention, age, type of hypospadias, details of surgical techniques used and outcomes as defined above.

Risk-of-bias assessment

The Cochrane Risk of Bias 2 (ROB2) tool was used to assess the methodological quality of included RCTs.[6] Domains assessed included the randomization process, Deviations from intended interventions, Missing outcome data, Measurement of outcomes, and Reporting bias. For non-randomized studies, we used the Risk Of Bias in Non-randomised Studies of Interventions (ROBINS-I) tool.[6] We used the Shiny app (robvis tool) for risk of bias assessment.[7]

Effect measures

Pooled effect estimates for dichotomous outcomes (e.g., complication rates) were calculated as risk ratios (RRs) with 95% confidence intervals (CIs). Continuous outcomes were reported as mean differences (MDs). Heterogeneity was assessed using the I² statistic. Thresholds: I² >50% indicated substantial heterogeneity.

RESULTS

A total of 272 records were retrieved from multiple databases – Embase (49), PubMed (41), Scopus (163), Cochrane (3), and other sources (16). One hundred and eighty-four of these were screened after the removal of 88 duplicate records. Of 184, 164 were excluded and 20 studies were retrieved for full text review. Finally, 10 studies[8,9,10,11,12,13,14,15,16,17] were found eligible to be included in quantitative analysis [Figure 1]. A study by Wei et al.[15] in Chinese had an abstract in English. We were able to extract all necessary information from the abstract except for the follow-up duration; hence, it was included in the quantitative analysis. The summary of all included study is mentioned in the study characteristic table [Table 1]. The outcome of the included studies and outcomes considered for quantitative analysis are mentioned in the outcome Table [Table 2].

Figure 1.

PRISMA flowchart for study selection

Table 1.

Baseline characteristics of participating studies

Authors Year, Setting	Study period	Study design	Patients (n) Total (CC, FR)	Age (Mean/Median, Range/IQR)	Type of hypospadias Procedure	Follow up period	Reported Outcomes
Cimador M et al.[8] 2003, Italy	January 1996- January 2001	Prospective	186 (CC-27, FR-159)	17 months	Distal penile Mathieu/MAGPI	3.7 years	UCF, dehiscence, stricture, phimosis
Suoub M et al.[9] 2008, Canada	September 2002- June 2007	Prospective	74 (CC- 49, FR-25)	13-15 months	Glanular, Coronal TIP	17-19 months	UCF, dehiscence, phimosis, operative time, redo – surgery
El Ganainy EO et al.[10] 2011, Egypt	2006-2009	RCT	153 (CC – 67, FR- 86) (200 patients recruited, 100 in each group, 153 completed follow up	CC- 35.2 (9-76) months FR- 35.2 (8-78) months	Distal penile/Coronal Mathieu	3 months	UCF, GD, preputial edema, mean operative time
Moslemi MK et al.[11] 2011, Iran	March 2004 – March 2009	RCT	86 (CC- 43, FR- 43)	Range: 1-11 years Mean: 7.9 years Median: 3 years	Mid/distal penile Mathieu	1 year	UCF, MS, transient preputial edema, preputial necrosis, cosmesis
Esposito C et al.[12] 2013, Italy	January 2007 - December 2011	Prospective	445 (CC - 91, FR - 354)	8-120 months	Glanular, Coronal, Subcoronal TIP in CC, MAGPI/TIP in FR	Up to 12 months	UCF, dehiscence, stenosis, HOPE score
Snodgrass W et al.[13] 2013, USA	September 2000-July 2011	Prospective Case Cohort	428 (CC – 343, FR – 85)	CC- 8 (4-12)* years FR- 8 (5-18)* years	Distal TIP	CC-7 (3-10) * FR- 8 (4-11) *	UCF, GD, MS, stricture, skin complications
Pan P et al.[14] 2020, India	January 2017- December 2019	RCT	80 (CC- 40, FR 40)	CC-4.59 +/- 1.43 years FR- 4.6 +/- 1.52 years	Distal/Mid penile TIP	12 months	UCF, GD, MS, Urinary Stream, FR - Wound gaping & dehiscence
Wei L et al.[15] 2020, China	November 2017-April 2018	Prospective	64 (CC- 36 FR 28)	Not mentioned in abstract	Distal TIP	Not mentioned in abstract	UCF, GD, MS, preputial dehiscence
Chhabra A et al.[16] 2022, India	Not mentioned	Prospective Comparative	50 (CC – 25, FR – 25)	CC- 5.58 (1.5-13) years FR- 4.06 (1-12) years	Distal, Subcoronal TIP	2 months	UCF, mean catheter duration, narrow meatus
Abdelwahab MM et al.[17] 2023, Egypt	December 2021- August 2022	RCT	40(CC- 20, FR- 20)	Less than 12 years	Distal TIP	Not mentioned	Early Complications (Edema, GD, preputial dehiscence, irregular preputial hood, infection), Late Complications (UCF, MS, stricture) Operative Time, Hospital Stay Parent Satisfaction

RCT – Randomised controlled trial, CC- Circumcision, FR – Foreskin Reconstruction *- median with IQR, TIP – Tubulerised Incised Plate urethroplasty, UCF- Urethrocutaneous Fistula, GD – Glans Dehiscence, MS – Meatal Stenosis, MAGPI – Meatal Advancement and Glanuloplasty Incorporated

Table 2.

Outcomes in the participating studies

Study	CC/FR	Total (n)	UCF	MS	GD	Skin/Preputial complications (Like dehiscence, necrosis )	Urethral Stricture	Mean Operative Time (mins)#
Cimador M et al.[8]	CC	27	1	2*	0	0	2*	-
	FR	159	8	11*	0	6	11*	-
Suoub M et al.[9]	CC	49	4	0	0	0	0	75 (35-113)
	FR	25	3	0	0	1	0	57 (27-86)
ElGanainy EO et al.[10]	CC	67	6	0	1	0	0	121.7 (87-167)
	FR	86	7	0	1	0	0	64.5 (49-82)
Moslemi MK et al.[11]	CC	43	5	1	0	0	0	-
	FR	43	6	1	0	1	0	-
Esposito C et al.[12]	CC	91	2	1	0	0	0	-
	FR	354	11	3	0	16	0	-
Snodgrass W et al.[13]	CC	343	16	1	13	7	0	-
	FR	85	4	0	2	2	1	-
Pan P et al.[14]	CC	40	2	1	1	0	0	-
	FR	40	3	1	0	1	0	-
Wei L et al.[15]	CC	36	1	1	2	0	0	143.8+/-3.0
	FR	28	1	1	0	2	0	109.9+/-2.2
Chhabra A et al.[16]	CC	25	2	3	0	0	0	-
	FR	25	1	4	0	1	0	-
Abdelwahab MM et al.[17]	CC	20	4	2	1	0	0	58.5 +/- 6.50
	FR	20	2	1	0	3	1	72.5 +/-8.35

CC- Circumcision, FR – Foreskin Reconstruction , UCF- Urethro-cutaneous Fistula , MS – Meatal Stenosis, GD – Glans Dehiscence, *combined outcomes of MS and Urethral Stricture , ^#blank rows , outcomes has not been reported

Urethrocutaneous fistula

All studies included in this quantitative analysis reported UCF as the outcome (total events: FR = 46/865, CC = 43/741). The pooled risk ratio (RR) for this outcome for all included studies was 1.06 [0.65, 1.74] [Figure 2]. This implies no significant difference in the incidence of UCF between FR and CC. No heterogeneity was observed among the included studies (I² = 0%) for this evaluation. Subgroup analysis of the RCTs which includes the four studies[10,11,14,17] also showed statistically non-significant results between these two groups (FR and CC) with RR 0.97 (0.52, 1.81) without heterogeneity. Exclusive Mathieu repair was done by ElGanainy et al.[10] and Moslemi et al.[11] A subgroup analysis for this group was statistically not significant among both groups with RR 1.03 (0.48, 2.20) without heterogeneity. Exclusive tubularized incised plate (TIP) repair was done in 6 studies in both groups.[9,13,14,15,16,17] Subgroup analysis of this group (TIP) also showed no statistically significant difference between both CC and FR without heterogeneity (RR 0.97 [0.49, 1.93]).

Figure 2.

Forest Plot showing pooled risk ratio for urethra-cutaneous fistula

Meatal stenosis

Eight studies were included for analysis of this outcome. A study by Cimador et al.[8] did not report MS separately but included it under urethral or meatal stricture in 11 patients; hence this study was excluded from the final analysis for this outcome. A total of 11 events were reported in the FR group and 10 in the CC group. The pooled RR was 1.02 [0.44, 2.38] [Figure 3] indicating no significant difference in MS risk between the two groups. Also, no heterogeneity was detected among the included studies (I² =0%), suggesting consistency in the results.

Figure 3.

Forest Plot showing pooled risk ratio for meatal stenosis

Glans dehiscence

Five studies included in quantitative analysis reported this outcome. The pooled RR was 0.50 [0.18–1.43], indicating no significant difference in GD risk between the groups. Heterogeneity was absent (I² =0%), suggesting consistency among included studies.

Skin/Preputial complication – Nine studies included in this analysis reported these complications under different categories, including preputial dehiscence, necrosis, and preputial edema. Since preputial edema is a transient complication, it was excluded from this category, and the remaining complications were grouped for the analysis. The pooled RR for this outcome was 2.89 [1.20–6.97], with no statistical heterogeneity, pointing to a significantly higher complication rate in the FR group compared to the CC group.

Operative time

Four studies[9,10,15,17] have reported this outcome; however, only Abdelwahab et al.[17] and Wei et al.[15] have provided data in the form of mean and standard deviation. Two other studies by ElGanainy et al.[10] and Suoub et al.[9] provided data using mean and range. A pooled analysis of the two studies with compatible data formats showed a MD of 10.05 [−313.73, 293.62], with substantial heterogeneity (I² =98%). The remaining two studies were not included in the quantitative analysis. Suoub et al.[9] and El Ganainy et al.[10] has reported a longer operative time for CC group compared to FR group.

Risk of bias assessment

Risk-of-bias assessment for RCTs was done using the ROB-2 tool. ElGanainy et al.[10] and Moslemi et al.[11] have an “overall” judgment of “some concerns,” while Pan[14] and Abdelwahab et al.[17] have a low risk of bias [Figure 4]. Risk of bias assessment for prospective studies was done with using the ROBINS-I tool. All other prospective studies except Snodgrass et al.[13] et al. have a low risk of bias across all domains (D1-D7). Snodgrass et al.[13] have some concerns in the domain of missing data [Figure 5].

Figure 4.

Risk of bias analysis for randomized controlled trials with the Risk of Bias-2 tool

Figure 5.

Risk of bias assessment for prospective studies with the Risk of Bias in Non-randomised Studies of Interventions tool

DISCUSSION

This meta-analysis provides a comparative evaluation of FR and CC in hypospadias repair, focusing mainly on postoperative complications. Though both techniques are commonly used by surgeons, the results of our quantitative analysis provide valuable insights into their relative merits and limitations. The analysis demonstrated no statistically significant difference in the incidence of UCF between the two groups. This indicates that the choice of foreskin management either reconstruction or circumcision, does not significantly influence the risk of this common complication. These findings align with existing literature suggesting that the occurrence of UCF is more likely influenced by factors such as the neourethral repair length, patient age, previous urethral repair,[18] and use of interposition tissue layer, etc., rather than the choice of foreskin management. Similarly, no significant difference in MS was observed between the FR and CC groups. This outcome also highlights that FR does not increase the risk of MS. The pooled analysis showed no significant difference in the risk of GD between FR and CC thus suggesting that both approaches provide comparable glans stability.

The analysis revealed a significantly higher risk of preputial complications in the FR group compared to the CC group without heterogeneity. The increased incidence of prepuce-related complications in the FR group may be attributed to the greater surgical complexity and tissue manipulation involved in FR. In FR, the vascular supply to the preputial flap may be compromised, leading to an increased risk of necrosis and dehiscence. Suturing techniques used in FR can create tension at the repair site, predisposing to wound breakdown. Based on this quantitative analysis, surgeons can counsel patients or parents to balance the advantages of FR against possible drawbacks.

Contrary to the common belief that FR might require a longer operative time due to the additional time required for preputial reconstruction[19] available data presents a conflicting picture. While only one study has reported longer operative times for FR, three others found CC to be more time-consuming. This discrepancy could be influenced by variations in surgical technique (e.g., few surgeons do not adopt a dorsal degloving for FR[9]) and surgeon’s experience. The high statistical heterogeneity (I² =98%) in the pooled analysis further suggests significant variability across studies. Both FR and CC are viable options for foreskin management during hypospadias repair, and the choice should be guided by cultural, religious, and personal preferences, as well as the surgeon’s expertise. While CC may be simpler and associated with fewer transient complications, FR offers the benefit of preserving a natural preputial appearance, which may be preferred by some families.

Secondary phimosis is a known long-term complication following FR after hypospadias repair. This issue typically manifests later, as preputial retraction is generally advised only after 4–6 weeks postoperatively. Studies with short follow-up durations, such as those by El Ganainy et al.[10] and Chhabra et al.[16] (2 months), may not have captured the incidence of this complication. Preputial narrowing often responds to local steroid application,[13] though a surgical intervention may be necessary for some.[12]

Randomization between the FR and CC groups may not be technically feasible, given the strong cultural influences and prevalent ritual practices. Snodgrass et al.[13] has also highlighted similar contextual aspects regarding procedural preferences. The presence of significant dimpling or pronounced dorsal asymmetry is also regarded as a contraindication for FR.[19] For this reason, we included all prospective studies alongside available RCTs in our quantitative analysis.

The included studies have clinical heterogeneity-few have included distal and mid-penile hypospadias repair for analysis while few have studied only distal hypospadias. FR can be easy in distal hypospadias but may be difficult in mid-penile hypospadias; likewise, it is not usually advocated in the proximal hypospadias so no study has included patients with proximal hypospadias. Two studies have done Mathieu repair while six have performed the TIP but there is no heterogeneity among the compared groups in individual studies.

Limitations

There was no uniform surgical procedure performed across the group-some patients underwent TIP, some a MAGPI, and some others a Mathieu repair. In addition, two studies included midpenile hypospadias repairs in their analysis, further increasing the heterogeneity of the pooled data. Of the 10 included studies, only four reported operative time as an outcome, making it challenging to draw a definitive conclusion.

CONCLUSION

This systematic review and meta-analysis found no significant differences in major complications such as UCF, MS, and GD between FR and CC during hypospadias repair. However, FR was associated with a significantly higher risk of preputial complications, probably attributable to the technical demands of the procedure and increased tissue manipulation in FR.

Conflicts of interest

There are no conflicts of interest.

Funding Statement

Nil.