# Adult Urethral Stricture Disease after  Childhood Hypospadias Repair

Authors: Shou-Hung Tang; Clarisa C. Hammer; Leo Doumanian; Richard A. Santucci
Journal: Advances in Urology
 (2008)

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## Abstract

Background. Adult patients with urethral stricture after childhood
hypospadias surgeries are infrequently discussed in the
literature. We report our experience in treating such patients.
Materials and Methods. A retrospective chart review was performed.
From 2002 through 2007, nine consecutive adult patients who had
current urethral stricture and had undergone childhood hypospadias
surgeries were included. All adult urethral strictures were
managed by a single surgeon. Results. Mean patient age was 38.9
years old. The lag time of urethral stricture presentation ranged
from 25 to 57 years after primary hypospadias surgery, with an
average of 36 years. Stricture length ranged from 1 to 17 cm
(mean: 10.3 cm). Open graft-based urethroplasties were performed
in 4/9 cases. Salvage perineal urethrostomy was performed in 2/9
cases. Another 3 cases chose to undergo repeat urethrotomy or
dilatations—none of these patients was cured by such treatment.
Complications included one urethrostomy stenosis and one urinary
tract infection. Conclusion. Urethral stricture may occur decades
after initial hypospadias surgery. It can be the most severe form
of anterior urethral stricture, and may eventually require salvage
treatment such as a perineal urethrostomy. Patients undergoing
hypospadias surgery should receive lifelong follow-up protocol to
detect latent urethral strictures.


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## Body

## 1. Introduction

Hypospadias is a common congenital abnormality occurring in 1/300 live
births, and is the most common congenital penile anomaly [1, 2].  Numerous
surgical techniques have been developed to correct this anomaly. However, no single
method is considered the standard of care, and they all share the common
complications of occasional urethrocutaneous fistula and urethral strictures. The
incidence of urethral stricture after hypospadias surgery in pediatric population is reported, and occurs in about 6.5% after
short followup [3].  On the other hand, there are few
reports dealing with urethral strictures in adults after they had hypospadias
surgery in childhood.  In the current
series, we described our experiences in 9 such cases, and review their
particular characteristics and suggested treatments.


## 2. Materials and Methods

A retrospective chart review was
performed from 2002 through
2007. Nine consecutive adult patients
who had current urethral strictures
and had undergone childhood hypospadias surgeries were included in this
study. All adult urethral strictures
were managed by a single
surgeon (RAS). The strictures in these
patients were all symptomatic and were documented by retrograde urethrography
(RUG). Information regarding hypospadias
repairs, previous urethral manipulations, presenting
symptoms, stricture length, definite
treatment, and short-term outcomes were obtained from medical records. In cases undergoing perineal
urethrostomy, we suggested the “side-to-side” technique, which comprised
longitudinal urethrotomy and everting the mucosal and submucosal layers of the
urethra to the incised skin. Graft-based urethroplasty using buccal mucosal
graft, in one-stage or two-stage repairs, was the preferred choice of formal
reconstruction.


## 3. Results

Mean patient age was 38.6 years old, and mean followup period was 1.9
years. All patients had their primary hypospadias surgeries between 1 and 12
years old. The lag time of the adult urethral stricture presentation ranged
from 25 to 57 years, with an average of 36 years between hypospadias surgery
and presentation to our clinic with urethral stricture.Four of the 9 patients (44%) presented with acute urinary retention, and
one of these patients developed acute renal failure due to prolonged urinary
retention, before stricture was diagnosed. The other associated complications included fistula in one case.  Only three of the 9 patients (33%) suffered
from lower urinary tract symptoms (LUTS) including decreased voiding stream,
spraying, dribbling, and nocturia. In one case, urethral stricture was
discovered when we evaluated unresolved urethrocutaneous fistulas (see Figure1).
Before transferred to our institute, 5/9 patients had undergone endoscopic
treatment for strictures, and 2/9 had failed open urethroplasties.Figure 1
Typical complex urethral stricture after childhood hypospadias repair, with a
distal penile location and complicating fistulae.Penile urethra was involved in
all cases, and bulbar urethra was involved in 5/9. The stricture length ranged
from 1 to 17 cm (mean, 10.3 cm) (see Figure2, Table 1)Table 1
Characteristics  of 9 adult urethral stricture patients with childhood hypospadias repair.
agelength (cm)treatmentrecurrence251Single-stage
dorsal-onlay urethroplasty with buccal mucosal graftno266Single-stage
dorsal-onlay urethroplasty with buccal mucosal graftno394Two-stage
urethroplasty with buccal mucosal graft in first stageno555Two-stage
urethroplasty with buccal mucosal graft in first stageno2617DVIU/dilatationsyes4813DVIU/dilatationsyes3715DVIU/dilatationsyes3615perineal
urethrostomyno5717perineal
urethrostomyYes, Y-V plastyFigure 2
Typical
retrograde urethrogram appearance of adult stricture after hypospadias
repair showing a long stricture sparing only the bulbar urethra.Open urethroplasties with buccal mucosal grafts were performed in 4/9
cases: two with single-stage repair and two with 2-stage repair with buccal
grafts (see Figure3).  Salvage perineal
urethrostomy was performed in 2/9 cases, usually in patients who did not wish
complex definitive urethral surgery (see Figure 4). The perineal urethrostomy
was planned to be permanent. Another 3 cases chose to receive repeated
endoscopic treatments (direct visual internal urethrotomy (DVIU) and/or
dilatations) although perineal urethrostomy or urethroplasty had been offered.Figure 3
First-stage urethroplasty with buccal grafts.Figure 4
Perineal urethrostomy, performed using our suggested “side-to-side”
technique.None
was cured by DVIU/dilatations. Four patients receiving open urethroplasty with
buccal grafts were free of stricture recurrence. Stenosis of urethrostomy
developed in one case and was successfully managed by a V-Y plastic technique.


## 4. Discussion

There is little literature mentioning adult urethral stricture in
hypospadias patients. Barbagli et al. published a series of 60 adults with previously
failed hypospadias repair [4], including 34 cases that underwent
treatment for urethral strictures. Their overall successful rate was 75% (83%
for one-stage repair, 68% for multistage repairs). It was evident that those
who needed multistage repair plans were at higher risk of failure because they
had more severe strictures and extremely poor quality native tissue than those
on whom the surgeons would consider risking a single-stage repair.


### 4.1. How are these Hypospadias Stricture Patients Different?

Adult stricture patients with previous hypospadias repair differ from a
usual population of stricture patients. First, they sometimes had no voiding complaints even when their
strictures were severe. Second, they had complicating problems seldom seen in
other stricture patients, including complete renal failure and urethral
fistula. Third, they have a poor quality
of tissue which requires more complex repairs such as first-stage Johanson
operations, with buccal grafts placed in the first stage, followed by second-stage
closure later. The associated
complications do represent a factor influencing the surgical strategy. However,
major determinants were the stricture length, availability of healthy tissue, as
well as surgeon’s own preference. In patients with long stricture and prominent
scarring, we suggested staged repairs if formal reconstruction was planned. Last, they often have such long and hopeless
abnormal anterior urethras, that is, by both patient and surgeon, it is
determined best to treat them expediently with simple perineal urethrostomy
instead of formal repair. In this way, reliable egress of urine can be
virtually guaranteed after a short 1-2 hour operation,
an option chosen by 5/9 (56%) of our cases. Our experience here exemplified that heroic measures were not always justified to
treat the severest urethral
strictures, and that perineal urethrostomy can be a gratifying option.


### 4.2. Healing in Adults and Children

While differences in wound healing ability between children and adults
are well described [5], little direct data is available on the
relative behavior of adult and childhood tissue in the urologic arena. Adult hypospadias surgery has been reported, and
may provide some insight into the pitfalls of complex reconstructive surgery in
the adult. For example, in a series of adults who underwent adult hypospadias
repair, redo operations had a worse outcome than primary cases [6]. They found that previous surgeries and
poor tissue quality attributed to higher failure rates. There was a significant
difference in terms of wound healing, infection, complication rates, and
overall success in adults compared to children. Increased surgical difficulty
and high failure rate after redo adult hypospadias surgery may be well applied
to adult urethral strictures such as seen in our population.


### 4.3. Incidence of Adult Urethral Stricture after
Childhood Hypospadias Repair

Urethral stricture is a
known complication following
hypospadias repair [7–9], but the true incidence is unknown. Some childhood hypospadias series do not
follow the patients long enough to report any strictures, and when series do
report strictures, they usually report them as acute events that occur while
the patient is still in childhood, not later as adults. A series by Duel et al.,
for example, showed a stricture incidence as high as 6.5% (38 of 582) after
pediatric hypospadias surgeries [3]. They demonstrated that strictures occurred
after a (mean) interval of 27 months. 79% of these pediatric urethral
strictures ultimately required open urethroplasty for correction, and they had
a 78% overall successful rate.The wide range of stricture length in our series was mainly affected by
original type of hypospadias. Patients undergoing repair for scrotal
hypospadias would have greater chance to have a subsequent longer urethral
stricture. Unfortunately, the exact type of original hypospadias cannot be
determined simply by gross appearance or by history in most cases.


### 4.4. How to Treat the Adult Stricture Patient

Repair of posthypospadias strictures in children has
been widely discussed by pediatric urologists. Modern series now favored single-stage,
two-stage buccal mucosal graft repair, or urethroplasties utilizing tunica vaginalis [10, 11]. We agree, and tend to offer two-stage buccal mucosal repairs
such as described by Johanson in adults. We also acknowledge that some of these patients have such extensive
disease, and little interest in a two-stage operation to fix the problem, and
thus are most appropriately treated with a perineal urethrostomy. Perineal urethrostomy was offered as
a second choice in addition to formal urethral repair. Comorbidity and previous
failed urethroplasty were the major factors influencing patients who accepted
perineal urethrostomy.


## 4.1. How are these Hypospadias Stricture Patients Different?

Adult stricture patients with previous hypospadias repair differ from a
usual population of stricture patients. First, they sometimes had no voiding complaints even when their
strictures were severe. Second, they had complicating problems seldom seen in
other stricture patients, including complete renal failure and urethral
fistula. Third, they have a poor quality
of tissue which requires more complex repairs such as first-stage Johanson
operations, with buccal grafts placed in the first stage, followed by second-stage
closure later. The associated
complications do represent a factor influencing the surgical strategy. However,
major determinants were the stricture length, availability of healthy tissue, as
well as surgeon’s own preference. In patients with long stricture and prominent
scarring, we suggested staged repairs if formal reconstruction was planned. Last, they often have such long and hopeless
abnormal anterior urethras, that is, by both patient and surgeon, it is
determined best to treat them expediently with simple perineal urethrostomy
instead of formal repair. In this way, reliable egress of urine can be
virtually guaranteed after a short 1-2 hour operation,
an option chosen by 5/9 (56%) of our cases. Our experience here exemplified that heroic measures were not always justified to
treat the severest urethral
strictures, and that perineal urethrostomy can be a gratifying option.


## 4.2. Healing in Adults and Children

While differences in wound healing ability between children and adults
are well described [5], little direct data is available on the
relative behavior of adult and childhood tissue in the urologic arena. Adult hypospadias surgery has been reported, and
may provide some insight into the pitfalls of complex reconstructive surgery in
the adult. For example, in a series of adults who underwent adult hypospadias
repair, redo operations had a worse outcome than primary cases [6]. They found that previous surgeries and
poor tissue quality attributed to higher failure rates. There was a significant
difference in terms of wound healing, infection, complication rates, and
overall success in adults compared to children. Increased surgical difficulty
and high failure rate after redo adult hypospadias surgery may be well applied
to adult urethral strictures such as seen in our population.


## 4.3. Incidence of Adult Urethral Stricture after
Childhood Hypospadias Repair

Urethral stricture is a
known complication following
hypospadias repair [7–9], but the true incidence is unknown. Some childhood hypospadias series do not
follow the patients long enough to report any strictures, and when series do
report strictures, they usually report them as acute events that occur while
the patient is still in childhood, not later as adults. A series by Duel et al.,
for example, showed a stricture incidence as high as 6.5% (38 of 582) after
pediatric hypospadias surgeries [3]. They demonstrated that strictures occurred
after a (mean) interval of 27 months. 79% of these pediatric urethral
strictures ultimately required open urethroplasty for correction, and they had
a 78% overall successful rate.The wide range of stricture length in our series was mainly affected by
original type of hypospadias. Patients undergoing repair for scrotal
hypospadias would have greater chance to have a subsequent longer urethral
stricture. Unfortunately, the exact type of original hypospadias cannot be
determined simply by gross appearance or by history in most cases.


## 4.4. How to Treat the Adult Stricture Patient

Repair of posthypospadias strictures in children has
been widely discussed by pediatric urologists. Modern series now favored single-stage,
two-stage buccal mucosal graft repair, or urethroplasties utilizing tunica vaginalis [10, 11]. We agree, and tend to offer two-stage buccal mucosal repairs
such as described by Johanson in adults. We also acknowledge that some of these patients have such extensive
disease, and little interest in a two-stage operation to fix the problem, and
thus are most appropriately treated with a perineal urethrostomy. Perineal urethrostomy was offered as
a second choice in addition to formal urethral repair. Comorbidity and previous
failed urethroplasty were the major factors influencing patients who accepted
perineal urethrostomy.


## 5. Conclusion

Urethral stricture can occur
decades after initial hypospadias surgery. Patient often have few voiding
complaints and can present with
severe complications. The stricture can be very extensive and may require salvage treatment such as a perineal urethrostomy. Two-stage urethroplasties with buccal
mucosal grafts can achieve good result when necessary. We suggest that patients
undergoing hypospadias surgeries should receive lifetime followup to detect
latent urethral strictures, and that research reports discussing stricture
after hypospadias repair include very-long-term followup data to determine the
exact incidence of this problem.



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*Source: 150315-2008-11-04.xml*