+ MODEL
Journal of Pediatric Urology (xxxx) xxx xxx
a
Chirurgie infantile, CHU de
Nantes, Nantes, France
b
Urologie, CHU de Nantes,
Nantes, France
c
Chirurgie infantile, Hopital
Necker Enfants Malades, Paris,
France
* Correspondence to: Sebastien
Faraj, Chirurgie infantile, CHU
de Nantes, 7 Quai Moncousu,
44000 Nantes, France
Sebastien.faraj@chu-nantes.
fr (S. Faraj)
Thomas.loubersac@chu-
nantes.fr (T. Loubersac)
Olivier.bouchot@chu-nantes.
fr (O. Bouchot)
Yves.heloury@aphp.fr
(Y. Heloury)
Marcdavid.leclair@chu-
nantes.fr (M.-D. Leclair)
Keywords
Hypospadias; Stricture; Bulbar
stricture; Transitional urology;
Pediatric urology
Abbreviations
BMG, Buccal Mucosa Graft; GA,
General Anesthesia; TIP, Tubu-
larized Incised Plate; UTI, Uri-
nary Tract Infection
Received 18 February 2021
Revised 12 October 2021
Accepted 1 November 2021
Available online xxx
https://doi.org/10.1016/j.jpurol.2021.11.001
1477-5131/ª 2021 Journal of Pediatric Urology Company. Published by Elsevier Ltd. All rights reserved.
Please cite this article as: Faraj S et al., Adults with previous hypospadias surgery during childhood: Beware of bulbar strictures, Journal
of Pediatric Urology,Downloaded
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Adults with previous hypospadias surgery
during childhood: Beware of bulbar
strictures
Sebastien Faraj a,*, Thomas Loubersac a,b, Olivier Bouchot b,
Yves Heloury c, Marc-David Leclair a
Summary
Background
Urethral strictures represent the most frequent
long-term complication in men after history of
hypospadias surgery.
Objectives
To better define the spectrum of men previously
treated for hypospadias during childhood, present-
ing at an adult urology clinic with persistent urethral
complications.
Study design
Retrospective review of the records of 42 consecu-
tive adult patients with a personal history of hypo-
spadias repair during childhood referred at one adult
tertiary urology center between 2004 and 2017.
Those with persistent urethral strictures were
included: 28 patients (median age 28yr [17e81]).
Residual chordee, aesthetic concerns, isolated fis-
tulas, and motives of consultation unrelated to
hypospadias were excluded. Early initial success
rate and location of urethral stricture were studied.
Statistical Analysis was done by non-parametric
tests.
Results
On the 42 consecutive adults eligible, a total of 28
patients had a persistent urethral stricture. During
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childhood, 13/28 boys had required multiple surgical
revision procedures, including six of them for post-
operative urethral strictures. During adulthood,
initial urethral assessment revealed 29 urethral
strictures in 28 patients (penile urethra 23/29,
bulbar urethra 8/29). The early initial success rate
of stricture treatment was 50% (median follow-up: 4
years [1e7]). Additional surgical procedures were
needed and performed in 11 patients. Eight patients
were eventually diagnosed with a bulbar stricture,
either isolated (n Z 5) or combined with a distal
stricture (n Z 3), without significant relationship
with initial position of meatus.
Discussion
Predictive factors for bulbar strictures locations
could not be identified. It was however observed
that symptomatic bulbar strictures do exist in
adults, in the long term after penile hypospadias
repair during childhood, independently of the initial
site of hypospadias, initial success of primary repair,
and the type of surgical reconstructions performed
during childhood.
Conclusion
Bulbar strictures represent more than 25% of the
overall strictures diagnosed in adult patients treated
for hypospadias during childhood, independently of
the original site of urethral reconstruction.
 + MODEL
1.e2
Summary Table Characteristics of patients according to stricture location.
n[
Initial position of meatus
Anterior
Penile
Posterior
Hypospadias first repair
MAGPI
TIP
Mathieu
Onlay
Duckett
Cecil-Leveuf
Late initial surgery (>3 years)
Staged primary surgery
Childhood complications after initial surgery
None
Stenosis
Fistula
Dehiscence
Other interventions during childhood
None
Non-urethral redo-surgery
Redo-urethroplasty and multiple surgeries
MAGPI: Meatal Advancement and GlanuloPlasty Intervention.
TIP: Tubularized Incised Plate.
Introduction
Hypospadias surgery is a challenge for paediatric urologists,
considering the wide range of clinical presentations and the
profusion of different surgical techniques. The long-term
follow up of hypospadias is a challenging and understudied
topic [1,2]. It is also a challenge for transitional urologists
who deal with long-term complications. Among these,
urethral strictures stand as the most frequent long-term
complications after history of hypospadias surgery during
childhood [3,4]. Other complications include urethro-
cutaneous fistulas, persistent chordee or aesthetic con-
cerns. Urethral stricture management in adult men remains
challenging, with several available surgical strategies and
inconsistent results. This is especially true in men with a
personal history of multiple stages of urethral reconstruc-
tion during childhood [5].
Our hypothesis was that urethral strictures can be
observed at any location along the urethra, and after any
technique of hypospadias repair, including in patients with
distal hypospadias repair. We conducted this retrospective
study to better define the spectrum of men previously
treated for hypospadias during childhood, presenting at an
adult urology clinic with persistent urethral complications.
Patients and method
We retrospectively reviewed the records of 42 consecutive
adult patients (Fig. 1) with a personal history of hypospa-
dias repair during childhood (1933e2000), referred at one
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S. Faraj et al.
DISTAL Stricture only BULBAR Stricture pZ
20 8
10 6 0.48
8 2 0,94
2 0 0,7
0 1 0,57
7 2 0,77
5 2 1
0 1 1
6 2 1
2 0 0,98
4 0 0,38
3 0 0,7
13 4 0,26
3 3 0,32
4 0 0,28
1 1 1
9 2 0,61
3 1 1
8 5 0,46
adult tertiary urology center between 2004 and 2017. Re-
quests were done on hospital software to combine hypo-
spadias diagnosis and urethral intervention. The software
used the ICD-10 (International Classification of Disease for
Mortality and Morbidity Statistics) and ICD-11 classification.
Asymptomatic patients with systematic routine follow-
up (n Z 8), patients referred for unrelated urologic con-
dition (n Z 3), and men with hypospadias-related penile
complication without urethral stricture (persistent isolated
urethrocutaneous fistula, residual chordee, aesthetic con-
cerns, n Z 3) were excluded.
Patients with enduring urethral symptoms (i.e dysuria,
episodes of urinary retention, pain, recurrent urinary tract
infections suggestive of urethral stricture, persistent ure-
throcutaneous fistula proximal to urethral stricture) were
included. Data regarding initial description of the malfor-
mation, demographic data, surgical procedures performed
during childhood and adulthood were recorded. Data were
collected from pre-operative, per-operative and post-
operative reports. Childhood was defined as pre-pubertal
period. We focused on postoperative outcomes of urethral
stricture surgery during adulthood. Urethral stricture was
confirmed by the association of stricture-compatible
symptoms and retrograde urethrogram or a flow-rate infe-
rior to 12 cc/sec [6]. Follow-up was calculated after the
last surgical procedure performed. Early success after
initial adult surgery was defined as absence of post-
operative urethral complication after a minimum follow-up
of 3 months. Redo-surgery was preceded by the persistence
of clinical symptoms or the persistence of stricture on the
retrograde urethrogram.
 + MODEL
Adults with previous hypospadias surgery 1.e3

procedure in 9 children (including two with foreskin

reconstruction), Duckett island-flap urethroplasty in 8,
Mathieu meatal-based flap procedure in 7, Cecil-Leveuf
(n Z 2), Onlay preputial island-flap (n Z 1), or MAGPI
(n Z 1). During childhood, 18/28 boys (64%) had required at
least one additional surgical procedure after initial repair,
including 13 who had already undergone multiple surgical
revision procedures before adulthood for urethral strictures
(n Z 6), persistent urethrocutaneous fistula (n Z 5) or
urethral distal dehiscence (n Z 2).

At adult age

Among the 28 patients, a majority of them (26/28, 93%)

presented with symptoms related to underlying outlet
obstruction (dysuria (n Z 19), episodes of acute urinary
retention (n Z 5), flank pain, recurrent febrile UTIs), or
urethra-cutaneous fistula (n Z 5). The reconstructed
meatus was described in hypospadiac position in 5 cases at
the first examination at adult age.
Initial endoscopic urethral assessment under anaes-
thesia revealed 29 urethral strictures in 28 patients (penile
urethra 23/29, bulbar urethra 6/29) associated with ure-
thral fistula upstream of the stricture in five cases and
urethral diverticulum in one patient. There were two pa-
tients with bulbar stricture not clearly identified at first
exploration but diagnosed later on, increasing the total
number of bulbar strictures to 8. No patient was diagnosed
with lichen sclerosus on physical examination.
First-line management of urethral stricture and imme-
diate outcomes are reported in Table 1, with a median
follow-up of 4 years [0e14]. Patients with bulbar stricture
were treated with internal urethrotomy (n Z 3), urethral
dilatation under GA (n Z 3), posterior skin flap ure-
throplasty (n Z 1) or direct end-to-end anastomotic repair
(n Z 1).
The early initial success rate of urethral strictures
treatment was 50%. Complication or recurrence was
observed in 14/28 patients, with a median delay of 4 years
Fig. 1 Flow-chart of the study.
[1e7], including 8 recurrent distal strictures and 5 bulbar
strictures in 11 men, fistulas (n Z 2), or stone on urethral
hair (n Z 1). Comparison between patients with early
Data were analyzed using R statistic software for non- successful versus failed stricture treatment showed no
parametric tests (Fisher exact test or Mann and Whitney
test), with p < 0.05 considered as significant.
Table 1 First line management of urethral stricture in
Results adulthood.
nZ Success Failure pZ
Twenty-eight men (median age 28 years [17e81]) with
Dilatation 9 4 5 ns
persistent urethral problems related to their hypospadias
Internal urethrotomy 4 3 1 ns
management during childhood were included.
Urethroplasty (flap) 9 4 5 ns
Posterior skin flap 5 2 3
During childhood De Sy meatal flap 4 2 2
Urethroplasty (graft) 4 2 2 ns
Among these 28 adult patients, initial hypospadias severity Buccal mucosa 1 1 e
could be retrospectively appreciated as distal in 16 (coronal Mesh graft 3 1 2
meatus without chordee), penile in 10 (mid-shaft meatus, Urethroplasty (other) 2 1 1 ns
with/without chordee), and proximal in two (peno-scrotal TIP 1 e 1
or perineal orifice). Urethrorrhaphy 1 1 e
Several different procedures had been performed as
TIP: Tubularized Incised Plate.
initial surgical repair: tubularized incised-plate (TIP)

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 + MODEL
1.e4
statistically significant difference in recurrence risk ac-
cording to initial position of meatus, association with se-
vere initial chordee, type or timing of initial repair, or
symptoms during adulthood. The outcome of adulthood
surgery was similar whether single or multiple urethral re-
constructions had been performed during childhood (9/14
vs 5/14, p Z 0.13).
Among the 14/28 patients with complications, three did
not need or consent to redo-surgery, including one treated
with auto dilatation (patient with double site strictures).
Additional surgical procedure was performed in 11 patients
(Table 2): redo-urethroplasty in one stage (n Z 7, including
four urethral reconstructions without graft or flap, two
mesh grafts and one posterior skin flap), redo-urethroplasty
and multiple stage revisions (n Z 3, performed with buccal
mucosa grafts), or single urethrotomy (n Z 1). After failed
repair of urethral stricture during adulthood and various
subsequent reconstruction procedures, 8/14 patients were
asymptomatic, but four had persistent symptomatic stric-
ture (2/4 performing urethral auto dilatation) and two had
urethrocutaneous fistula, after a median follow-up of 5
years [1e14].
A total of 8 patients were eventually diagnosed with a
bulbar stricture, isolated (n Z 5) or combined with a distal
stricture (n Z 3), either recognized at first examination or
after failed initial management. Characteristics of these
patients and comparison with men with distal stricture are
detailed in Table 3, without demonstrable features pre-
dictive of the occurrence of a proximal stricture. The initial
position of meatus, presence of associated chordee and
type and timing of primary urethroplasty did not seem to be
associated with a higher risk of bulbar stricture. However, it
appeared that 6/8 (75%) of patients presenting during
adulthood with a bulbar stricture had a history of multiple
urethroplasty procedures during childhood.
Discussion
The present study highlights the possibility of urethral
stricture in adults in the long term after hypospadias repair
during childhood, even in those who had undergone un-
eventful single primary repair. It also demonstrates an un-
expected proportion of bulbar strictures as sequelae of
previous surgical procedures and stresses the technical
challenge of stricture management during adulthood.
It is widely accepted that hypospadias surgery remains a
technical challenge [7,8]. Barbagli et al. reported a large
series of 1176 failed hypospadias in adult patients, in whom
a median of three operations were required to achieve
successful repair of a primary hypospadias [4]. The need for
long term follow-up after hypospadias repair has more
recently been recognized, considering the possible deteri-
oration of urethroplasty with time, even with apparently
excellent outcome in childhood [1,9]. One hypothesis could
be that congenital lack of spongiosum may contribute to
urethral deterioration, especially as a consequence of
sexual activity and repeated erections [3,4]. CB Ching et al.
reported that adult patients requiring redo-urethroplasty
after history of hypospadias represented a heterogenous
group, and fell into two distinct categories: most patients
(32/48, 67%), experienced a long and continuous history of
failed multiple surgical reconstructions, but a significant
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S. Faraj et al.
subgroup of adults (33%) presented with late failure during
adulthood after an initially successful childhood repair [10].
Half of the 28 patients of the present series reached
adulthood with an impression of acceptable result of their
malformation surgical repair and presented with late
complication, regardless of the number of surgical pro-
cedures required during childhood. It is also noteworthy
that several adult patients delayed the moment to seek
management of a long ongoing problem. This could be
related to the psychological consequences and psycho-
sexual burden of their personal surgical history, or in the
unawareness of the need for urological support in the
transition from adolescence to adulthood [11,12].
Hypospadias complications leading to adult urologist
consultation typically include stricture, cosmesis dissatis-
faction, abnormal position of the meatus, urethrocuta-
neous fistula, and persistent penile curvature [11,13]. As
expected, the main complication observed in the present
study was persistent urethral strictures in more than 90% of
the patients. It is well known that in the adult population, a
significant proportion of penile urethral strictures origi-
nates from hypospadias surgery during childhood [14].
Recruitment biases may explain the variable incidence of
hypospadias patients among adults with urethral strictures.
In a series of 125 adults with urethral strictures, V. Zum-
stein et al. reported 38% iatrogenic causes, 21% traumatic
or inflammatory, but also 24% after hypospadias surgery
[15]. Similarly, N. Lumen et al. identified hypospadias as a
leading aetiology, accounting for 20% of strictures in men
before 45 years [16]. However, urethral stricture aetiology
remains unclear and classified as idiopathic/unknown in
approximatively 30%e50% of adult cases [16,17]. G. Bar-
bagli et al. showed that the vast majority of urethral
strictures occurred at the original site of primary hypo-
spadias surgery [18], and described bulbar location in less
than 10% of their cases. In the present series, we observed a
surprisingly high rate of bulbar strictures (8/28), including
three patients with bifocal bulbar and distal strictures. N.
Hoy reported two cases of bulbar location in a series of
adult men with urethral stricture after hypospadias recon-
struction [19]. If distal urethral stricture can be viewed as a
direct complication of urethral surgical reconstruction, it is
more obscure to elucidate what drives the appearance of a
bulbar stricture in a male urethra which underwent distal
urethral surgical interventions several years before. Several
explanations have been proposed for so-called idiopathic
bulbar stricture, including childhood unrecognized trauma,
congenital origin, infectious, or ischaemic. Adult men with
history of multiple surgery for hypospadias share several of
these predisposing factors. Hence, it appears that men with
history of hypospadias are indeed over-represented in se-
ries of patients with bulbar strictures [16]. Repeat tran-
surethral manipulations, especially peri-operative stenting,
or use of tourniquet may have played a role, although one
could expect these traumatic strictures to be diagnosed
sooner. Another hypothesis is that bulbar stricture could be
a long term evolution of a subclinical outflow obstruction at
the site of prior surgery and a subsequent urethritis. Our
data did not allow to isolate specific factors predictive of
bulbar location, beyond a trend to history of multiple ure-
thral reconstructive procedures. Even if unrelated to
hypospadias history, it is interesting to observe that bulbar
 Adults with previous hypospadias surgery
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Table 2 Patients with early failure of stricture repair.

Patient CHILDHOOD COMPLICATIONS FOLLOW UP
Initial type of Hypospadias Other Age at first Stricture Procedure Type of Complication Follow-up Outcome
hypospadias first interventions adult surgery location at first adult complication management (yrs)
procedure during surgery
childhood
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https://doi.org/10.1016/j.jpurol.2021.11.001

1 Anterior MAGPI None 22 Bulbar Dilatation Bulbar stricture Bulbar 4 Asymptomatic

ureterorrhaphy
2 Anterior Duckett Multiple 19 Bulbar Dilatation Bulbar stricture Buccal mucosa: 8 Asymptomatic
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bulbar
urethroplasty
(x2)
Internal
urethrotomies
(x6)
3 Anterior Matthieu Multiple 28 Bulbar Posterior skin Distal and Buccal mucosa 6 Asymptomatic
& Distal flap bulbar for bulbar and
urethroplasty strictures distal
urethroplasties
4 Penile Duckett None 26 Distal Dilatation Distal stricture Observation 1 Asymptomatic

+
MODEL
5 Penile TIP Multiple 33 Distal Dilatation Distal and Autodilatation 12 Dysuria
bulbar
strictures
6 Anterior Matthieu None 81 Distal De Sy Distal stricture Observation 4 Asymptomatic
urethroplasty
7 Penile Duckett Multiple 64 Distal Posterior skin Distal stricture Dilatation 1 Asymptomatic
flap under general
urethroplasty anesthesia
8 Anterior Matthieu Multiple 46 Distal Posterior skin Fistula De Sy 1 Fistula
flap urethroplasty
urethroplasty
9 Anterior Matthieu Multiple 44 Distal Dilatation Stone on hairy Redo 1 Dysuria
urethra urethroplasty
with ablation of
stone on hairy
urethra
10 Anterior Matthieu Skin Z plasty 20 Distal TIP Fistula Posterior skin 2 Dysuria
for webbed flap
penis urethroplasty
11 Anterior Onlay Redo 67 Distal Internal Bulbar stricture Perineal 1 Asymptomatic
tubulization urethrotomy urethrostomy
12 Anterior TIP Multiple 23 Distal De Sy Distal stricture Meatoplasty 6 Dysuria

1.e5
urethroplasty
(continued on next page)
 Table 2 (continued )

1.e6
of Pediatric Urology,
Please cite this article as: Faraj S et al., Adults with previous hypospadias surgery during childhood: Beware of bulbar strictures, Journal

Patient CHILDHOOD COMPLICATIONS FOLLOW UP

Initial type of Hypospadias Other Age at first Stricture Procedure Type of Complication Follow-up Outcome
hypospadias first interventions adult surgery location at first adult complication management (yrs)
procedure during surgery
childhood
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Downloaded

13 Penile Cecil-Leveuf Dilatations 35 Distal Mesh Graft Distal stricture Posterior 9 Asymptomatic
https://doi.org/10.1016/j.jpurol.2021.11.001

sessions urethroplasty skinflap

urethroplasty
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Internal
urethrotomy
Mesh graft
urethroplasty
(two steps)
14 Posterior Duckett Multiple 39 Distal Mesh Graft Distal stricture Internal 14 Fistula
urethroplasty urethrotomies
(x6)
Mesh graft
urethroplasty
(two steps)

+
MODEL
MAGPI: Meatal Advancement and GlanuloPlasty Intervention.
TIP: Tubularized Incised Plate.

S. Faraj et al.
 + MODEL
Adults with previous hypospadias surgery 1.e7

previously treated for hypospadias repair, and identified

Table 3 Characteristics of patients according to stricture
the length of stricture and associated presence of lichen
location.
sclerosus as independent factors for adverse outcom [18].
DISTAL BULBAR pZ In a similar study, JB Myers et al. reported a 50% initial
Stricture Stricture success rate, rising to 76% after multiple additional pro-
only cedures [5]. Endoscopic management seems to have
n[ 20 8 disappointing results as first-line treatment; however, it
Initial position of meatus offers the advantage of a complete urethral evaluation, to
Anterior 10 6 0.48 ascertain precise location of the stricture.
Penile 8 2 0,94 Limitations in this study included potential bias in the
Posterior 2 0 0,7 recruitment pattern of these adult patients [24], and the lack
Hypospadias first repair of knowledge of the denominator (namely the number of
MAGPI 0 1 0,57 hypospadias patients treated during childhood). Unavailable
TIP 7 2 0,77 medical detail in ancient operative records did not allow
Mathieu 5 2 1 drawing a detailed picture of the surgical history of some
Onlay 0 1 1 patients and prevented to identify predictive factors for
Duckett 6 2 1 bulbar strictures locations. Presence of catheter or use of the
Cecil-Leveuf 2 0 0,98 tourniquet would be two hypotheses for the occurrence of
Late initial 4 0 0,38 bulbar stricture after hypospadias repair, that would require
surgery animal experimental histological studies for validation.
(>3 years) Length and exact location of bulbar strictures were not dis-
Staged primary 3 0 0,7 cussed because of absence of data in some patients. It was
surgery however observed that symptomatic bulbar strictures do
Childhood complications after initial surgery exist in adults, in the long term after penile hypospadias
None 13 4 0,26 repair during childhood, independently of the initial site of
Stenosis 3 3 0,32 hypospadias, initial success of primary repair, and the type of
Fistula 4 0 0,28 surgical reconstructions performed during childhood.
Dehiscence 1 1 1
Other interventions during childhood
None 9 2 0,61 Conclusion
Non-urethral 3 1 1
redo-surgery Urethral stricture is the most common complication in men
Redo- 8 5 0,46 with history of hypospadias repair. Surgical management of
urethroplasty these strictures remains a challenge, with an initial
and multiple reconstruction success rate of 50%. More than 25% of these
surgeries strictures are located at the level of bulbar urethra, war-
MAGPI: Meatal Advancement and GlanuloPlasty Intervention. ranting a comprehensive examination of the whole urethra
TIP: Tubularized Incised Plate. in symptomatic men even after distal hypospadias surgery.

strictures can be overlooked in this context and belatedly

recognized.
Various techniques of stricture repair have been
described in adult surgery, such as urethroplasty based on
penile skin flaps, primary excision and anastomosis, MESH
and buccal mucosa grafts (BMG) in a single or staged repair.
A recent observational study of multi-institutional surgical
trends suggested a migration away from urethral transec-
tion and ventrally placed grafts toward nontransecting re-
pairs whenever feasible and dorsal grafting [17]. In the
specific context of failed hypospadias surgery, the presence
of scarred penile skin and urethra and impaired vascular
supply pushes to techniques importing supplementary
healthy tissue, typically a buccal mucosa graft in one or
two-stage strategies [20e22]. Meeks et al. achieved a 86%
success rate using a multi-stage approach with BMG [23]. In
the present series, the initial success rate of primary ure-
throplasty for urethral stricture was 50%. This is similar to
previously reported results in larger series of adults: G.
Barbagli et al. showed a 5-yr failure-free survival rate of
48% in a series of 408 adult patients with strictures
Funding
This research did not receive any specific grant from
funding agencies in the public, commercial or not-for-profit
sectors.
Availability of data and material
All data and material are available and in the possession of
the corresponding author.
Ethics approval
This study was performed in line with the principles of the
Declaration of Helsinki in lieu of formal ethics committee
approval. This is a retrospective study and all the proced-
ures being performed were part of the routine care.

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 + MODEL
1.e8
Consent to participate
Informed consent with guarantees of confidentiality was
obtained from all individual participants included in the
study.
Conflicts of interest
The authors have no conflicts of interest to declare that are
relevant to the content of this article.
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