Failed Hypospadias in Paediatric Patients
Failed Hypospadias in Paediatric Patients
Failed Hypospadias in Paediatric Patients
Introduction
Hypospadias is a spectrum disorder. Meatal location Accordingly, reported reoperation rates after primary
and associated ventral curvature are the two major procedures can exceed 50%.2 Failed hypospadias occurs
variables that determine the severity of the condition, when the primary surgery does not achieve the cosmetic
ranging from patients with a glanular meatus and no and functional goals of a straight penis with a glanular
associated curvature to those with a perineal meatus meatus that enables normal urinary and sexual function.
and associated curvature of >90°.1 Preoperative assess- Failed hypospadias includes all complicated repairs, as
ment of hypospadias severity is often misleading. The well as uncomplicated repairs that do not fulfil patient
actual starting point of the malformation (where the expectations and cause dissatisfaction. It is worth noting
spongiosum splits into two halves leaving a thin urethra that none of the repairs available to date can be used
in between) is often much more proximal to the visible to create a urethra with the same biological and uro
hypospadic meatus and its exact location can only be dynamic properties as a native urethra and, therefore,
Section of Paediatric
determined after skin degloving. Furthermore, the functional outcomes, particularly voiding and ejacu- Urology and Paediatric
manoeuvres required for penile straightening can be latory function, can be severely affected even in the Surgery Unit,
Department for Mother
very different in patients with comparable degrees of absence of any complications. 4 Furthermore, patient and Child Care and
curvature and can only be determined intraoperatively satisfaction is very subjective and mainly depends on Urology, University
of Palermo,
using a step-wise approach, going from ventral dissec- the ability of the patient to cope with cosmetic and func- Via A. Giordano 3,
tion (with or without dorsal plication) to urethral plate tional abnormalities. Patient perception of the outcomes 90127 Palermo, Italy
transection (with or without procedures for lengthen- can, therefore, be very subjective and diverge widely (M. Cimador,
E. De Grazia).
ing the ventral radium of the penis).2 Other anatomical from the assessment of the surgeon. For example, the Paediatric Urology Unit,
characteristics that can influence technique selection prevalence of persistent curvature after hypospadias Ospedale
Maggiore‑Policlinico,
and surgical success include the width of the urethral repair was significantly lower in patients for whom Via Commenda 10,
plate, the depth glans, and the overall size of the penis.2 curvature was assessed by the patient or a parent com- 20122 Milan, Italy
In general, when the penis is shorter, ventral lengthen- pared with the surgeon (presumably because these (S. Vallasciani,
G. Manzoni). Section of
ing procedures are favoured over dorsal shortening ones, individuals do not give the same relevance to the same Paediatric Urology,
and the patient is more likely to be dissatisfied in the degree of curvature),5 and adolescents with hypospadias Urology Unit, University
Hospital of Padova,
absence of complications. seems to be generally less satisfied with the long-term Via Giustiniani 2,
More than 300 surgical procedures have been results of a repair than the paediatric urologists who 35128 Padua, Italy
described for hypospadias repair, suggesting that no operated on them as children.6 (W. Rigamonti,
M. Castagnetti).
single approach is fully satisfactory for all variants. 3 The burden of a failed hypospadias repair can be
devast ating for a patient and his family. In a series Correspondence to:
M. Cimador
Competing interests of 1,176 patients with failed hypospadias, a median of marcello.cimador@
The authors declare no competing interests. five additional procedures (ranging from 2–23) were unipa.it
is probably the most important surgeon-independent Box 1 | Factors influencing results in hypospadias repair
factor. Wound-healing processes are strongly influ-
Factors related to the patient
enced by hormones, and sex steroids have contrasting
■■ Endocrine environment
roles in the regulation of cutaneous repair processes.36,37 ■■ Wound-healing impairment
Oestrogens promote wound retraction by enhancing ■■ Decreased growth factor activity
dermal repair processes, inhibiting the production of Factors related to the surgeon
inflammatory cytokines, promoting re-epithelialization ■■ Expertise
and neoangiogenesis, and increasing the production ■■ Choice of procedure
of platelet-derived growth factors. Androgens, on the ■■ Perioperative and postoperative management
other hand, are thought to repress cutaneous repair in Factors related to the diagnosis
both acute and chronic situations, retarding the healing ■■ Incorrect identification of the urethral ending
process and increasing inflammation. These effects ■■ Incorrect evaluation of the urethral plate
might explain why some investigators have reported ■■ Incorrect evaluation of the spongiosum
poor outcomes in patients with hypospadias treated with Factors related to the procedure
androgen stimulation before urethroplasty.38 ■■ Inadequate technique
Furthermore, some studies39 have reported an imbal- ■■ Inadequate choice of repair
■■ Influence of suture materials
ance in the proportion of structural tissue-building epi-
■■ Urethral stenting
thelial tissue factors (such as cadherin E and claudin 1) to
destructive epithelial tissue factors (for example, metallo
proteinase 2) in the ventral penile tissue, but not the dorsal Factors related to the procedure
penile tissue, of patients with hypospadias. Perhaps this Technical details
could explain why better results were achieved using the Technical details are key to the success of hypospadias
onlay procedure, for which dorsal tissue is transposed ven- surgery. Coverage of the urethroplasty with multiple
trally. In this technical repair, the inner dorsal preputial layers of well-vascularized tissue is particularly important
layer is harvested as a flap, preserving the blood supply. for both distal and proximal hypospadias.47–49 The flaps
The harvested flap is then used to cover the native urethral most commonly used for uethroplasty coverage include
plate, thus providing the ventral wall of the neourethra. flaps of subcutaneous dartos elevated from around the
urethral plate, the prepucial pedicle transposed ven-
Factors related to the surgeon trally, and the tunica vaginalis flap. The importance of
Selection of the technique suture materials is unclear. Suture materials with a longer
Most urologists agree that no single procedure is appro- reabsorpt ion time might favour healing and prevent
priate for all hypospadias repairs1 and several surveys fistula formation, but they might also cause prolonged
have shown a wide variability in procedure choice among tissue reaction, potentially increasing the risk of stricture
different surgeons.40,41 Moreover, many surgeons have formation in the reconstructed urethra. Some studies
modified their surgical approaches as their practice has have indicated a key role in repair success,50,51 whereas
evolved.27,42 Very few studies have provided comparative others have shown no difference in urethral complication
data for different repairs performed in a uniform setting.43 rates when using different suture materials.52,53
Although some techniques have been associated with
some specific complications—for example, urethral diver- Postoperative management
ticula occurs almost exclusively after flap tube repairs44— In one study, early mobilization (within 48 h post
a systematic review of the literature on the treatment of operatively) was shown to decrease the incidence of fis-
primary severe hypospadias concluded that no single tulas from 22% to 9.8%, whereas factors such as catheter
urethroplasty technique is clearly superior to the others.2 dislodgement and blockage, presence of postoperative
erections, constipation-related straining, and inter
Surgeon expertise ference with dressings were associated with increased
In a recent ‘round table’ meeting of experts on hypo- complication rates.54 However, type and duration of
spadias, it was suggested that intellectual interest in the urinary diversion, type of dressing, catheter size, and
condition and number of operated cases are the two most anaesthetic regime have been shown not to significantly
important surgeon-dependent variables for predicting influence outcomes,55 and a recent review of the litera-
the success of hypospadias repair. 22 In a series of 299 ture on perioperative management of primary severe
patients undergoing primary hypospadias correction, hypospadias concluded that most interventions, such
complication rates improved considerably as the surgeon as postoperative antibiotic prophylaxis or wound dress-
became more experienced.45 A cut-off threshold of 100 ing,56–58 are based on weak evidence and their influence
cases per year has been proposed for classifying surgeons on repair outcomes is poorly defined.23 Only postopera-
as experts in hypospadias surgery,22 although one team tive urinary drainage has been consistently shown to be a
has suggested that this threshold should be lowered to 50 critical factor in reducing the complication rate.59
cases per year, as risk for hospital readmission within the
12 months after primary repair was found to be lower in Management of complications
centres treating more than 50 cases per year compared Failed hypospadias is not only a urethral issue; the
with those performing fewer procedures.46 corpore al bodies and penile skin are also commonly
involved. The most common complications include (Figure 1).61 Incorporation of additional extragenital
recurrent curvature, preputial dehiscence, glans dehis- tissue is often necessary for repair in these patients.
cence, urethral fistula or breakdown, meatal or urethral
stenosis, urethral stricture, urethral diverticulum, hairy Residual and recurrent curvature
urethra, penile skin deficiency, and abnormal penile Curvature can persist after a primary repair (owing to
skin configuration (Table 1). In a series of 50 adults with inappropriate correction during initial surgery) or recur
failed hypospadias, urethral strictures (n = 36), urethro if previous manoeuvres or attempts at ventral tethering
cutaneous fistulas (n = 12), persistent hypospadias (n = 7), have failed, leading to retraction of the reconstructed
hair in the urethra (n = 6), and severe penile curvature urethra or the penile skin. No longitudinal cohort studies
(n = 7) were observed.8 Complications were rarely iso- have been set up to determine the prevalence of curva-
lated. In a large series of failed hypospadias repairs, half ture persistence or recurrence in relation to the initial
of all patients undergoing urethral reconstruction also straightening manoeuvres. In a study of 100 patients with
required complex procedures to resurface the penile proximal hypospadias and severe curvature, recurrent
shaft or reconfigure the whole genitalia.60 When com- curvature was reported in a total of 22 patients, including
bined with urethral breakdown or stricture, scarring and three (of 32) children who underwent ventral lengthen-
retraction of the neourethra are likely to lead to recur- ing procedures and 19 (of 68) who underwent dorsal pli-
rent curvature. The term ‘hypospadias cripple’ is used cation (9% versus 28%; P = 0.03).6 This study suggests that
to describe a scarred hypovascular and shortened penis ventral lengthening could be more effective and durable
caused by multiple failed attempts at hypospadias repair in achieving penile straightening. However, in another
a b c
Figure 1 | A case of hypospadias cripple in a 19-year-old boy; the term is used to describe a scarred hypovascular and
shortened penis caused by multiple failed attempts at hypospadias repair. a | Persistent curvature during spontaneous
erection associated with skin scarring. b | Metallic probes show the presence of multiple urethral fistulas. c | Hairs are
detected inside the lumen of the scrotal urethra after opening.
study of 22 patients with recurrent curvature >10 years for dehiscence and 6.3% for secondary phimosis in
after initial penile straightening, 19 patients had received patients who underwent distal hypospadias surgery. After
dorsal plication as their initial procedure and three had a mean follow-up duration of 3.7 years, 90% of patients
undergone ventral tunica vaginalis grafting.16 Median had retractable foreskins.66,67 Although the relevance of
age at onset of recurrence was 16 years (ranging from cosmetic appearance following preputial reconstruction
12–18 years). These data show that penile curvature can remains controversial,68 cosmetic appearance was consid-
recur after both dorsal plication and ventral procedures, ered to be ‘good’ (according to surgeon assessment) for
and that most recurrences occur during puberty. all of our uncomplicated patients and none of the parents
Diagnosing residual curvature is not always easy, of these patients reported dissatisfaction.66 Overall, pre-
owing to the lack of a standardized diagnostic tool for putial reconstruction can be performed safely and with a
defining the degree of residual curvature that should low complication rate in appropriately selected patients.68
be considered clinically relevant. The surgeon often
has to rely on patient or parental report, which can Glans dehiscence
vary significantly from surgeon assessment.6 If physical Prevalence of glans dehiscence ranges from 0–8%. In the
examination during erection is unrealistic, photographs only specific study of this complication, risk of dehis-
should be taken by the patient during erection. In any cence was not affected by age at surgery, preoperative
case, if the correction of residual or recurrent curvature testosterone use, or glansplasty suture. However, the
is elected, an intraoperative erection test is mandatory risk of glans dehiscence after tabularised incised plate
before embarking on any further procedure. Treatment repair was 3.6-fold greater in patients with proximal
of residual or recurrent curvature should follow the same hypospadias compared with distal meatal hypospadias
principles as for primary curvature. 2 Minor residual and 4.7-fold greater for repeat procedures (14%) than for
curvature (<30°) can be left untreated provided it does primary repairs (4%).69 Most patients are treated conserv-
not interfere with sexual activity. If treatment is required, atively for glans dehiscence. Indeed, although a coronal
options include dorsal shortening procedures or ventral regression of the meatus is likely to have a negative effect
lengthening, which can sometimes require the removal on cosmetic appearance, this complication might actually
of all ventral scarred tissue and urethral substitution. improve urinary function, as it reduces outflow resist-
The former is potentially easier, but ventral lengthening ance and, therefore, the risk of urinary obstruction.
should be considered when the curvature is caused by a Indications for the correction of glans dehiscence are
shortage of skin or a contraction of the neourethra. similar to those for extremely distal hypospadias, namely
spraying, deviation of urinary stream, and unsightly
Inappropriate healing glans appearance.70 Several techniques can be used for
Preputial dehiscence and secondary phimosis repeat reconstruction of the distal urethra, including the
Preputial reconstruction is often requested by parents, well‑established meatal-based flipped flap approach.71
particularly in European countries with large numbers
of uncircumcised males. Moreover, the foreskin is erog- Fistula formation
enous tissue that could be important for the future sexual Reportedly, fistula formation occurs in 4–28% of patients
life of the patient.62–64 Unfortunately, preputial recon- after hypospadias repair,72 either in isolation or in associ
struction increases the risk of postoperative morbidity ation with a meatal or urethral stenosis. The presence of
and, therefore, of repair failure. Specific complications stenosis should always be ruled out before embarking on
include foreskin dehiscence and secondary phimosis. In fistula closure. Treatment depends on the number, loca-
one study, total dehiscence and phimosis were reported tion, and size of the fistulas. Prolonged catheterization
in 6% and 12.6% of patients treated with preputioplasty might be suitable for treating small fistulas that occur
for hypospadias repair.65 We have reported rates of 3.8% soon after the repair, whereas formal surgical repair is
Failed hypospadias: urethral breakdown urethral plate for reuse and the genital skin for mobili-
zation and incorporation in the new repair procedure
Curvature greater than 30° or interfering with sexual activity (Figure 2). When a suitable urethral plate is available,
possible strategies include tubularization of the plate
Yes No
(with or without a midline-releasing incision), tubu-
Residual urethra suitable to be incorporated in the re-urethroplasty larization of the urethral plate after inlay grafting of the
Yes
dorsal midline incision, and placement of a flap or graft
No
onlay over the urethral plate.76–78
Residual urethra sufficient to accomplish the re-urethroplasty When the plate is visibly scarred or deemed to contrib-
Yes No ute significantly to the persistence of ventral curvature,
urethral substitution is required, typically involving prepu-
Quality of genital tissue good enough
to mobilize a skin flap tial island tube placement or staged-graft urethroplasty.76,78
If extragenital tissue is to be used as graft owing to paucity
Yes No
of local tissue, most surgeons prefer to use an oral mucosa
Substition Tubularization of Flap Onlay or inlay graft harvested from the lower lip or inner cheek.79–81 The
urethroplasty the urethral plate urethroplasty graft urethroplasty
tongue and the retroauricular region are additional poten-
Figure 2 | Algorithm for the management of failed hypospadias owing to tial harvesting sites.82,83 No graft is definitively superior to
urethral breakdown. the others in terms of harvesting site morbidity or success
of the urethroplasty.84,85 Irrespective of the graft used,
a b
the first stage of repair involves quilting the graft onto
the ventral aspect of the penile shaft after the removal of
any scarred tissue. The graft is then tabularised around
6 months later. This staged approach to repair enables
the graft to ‘take’ properly before tabularization, which
minimizes the risk of postoperative complications.86
Urethral obstruction
Meatal stenosis
* Major causes of obstructed voiding after hypospadias
repair include meatal stenosis and urethral stricture.
* Meatal stenosis is particularly common with repair tech-
niques such as tabularized preputial flap urethroplasty
(performed when the glans is tunnelled) and tubularized
incised plate urethroplasty. The latter technique—which
Figure 3 | Retrograde and voiding urethrographies in a patient with urethral stricture
following hypospadias repair. a | During retrograde injection of contrast from the
is one of the most commonly performed repairs—is
glanular urethral meatus (denoted by an arrow), a narrow urethral segment is associated with a 0–14% risk of meatal stenosis, usually
visible. This segment, which is proximal to the stenosis, is dilated (denoted by an occurring within 6 months of the procedure.87 A deep
asterisk). b | During voiding, the dilated segment becomes even more apparent incision of the plate and the creation of a wide meatus
(denoted by an asterisk). No contrast is visible between the dilated urethral minimize the risk of this complication and negate the
segment and the tip of the penis (denoted by an arrow). need for calibration of the neourethra. 87 The most
common presenting symptoms are a weak stream, a
required for those that persist for >6 months after the long and thin stream, and spraying. Clinical diagnosis
initial hypospadias surgery. The repair can be simple or is generally based on meatal calibration; radiological
with flaps. Although simple repairs are easier, they are investigations are unnecessary unless proximal exten-
also less effective and, therefore, generally less appropriate sion of the stricture is suspected. Treatment is relatively
for large and recurrent fistulas.72–74 Redo distal urethro- straightforward and entails a ventral cutback of the
plasty might be more effective than fistula repair for fistu- external urethral meatus, namely a ventral meatotomy.
las that arise at, or distally to, the coronal sulcus. Urethral
mobilization and advancement might also be considered Urethral stricture
for treating persistently recurrent hypospadias fistulas.75 The reported prevalence of urethral strictures proximal
to the meatus after hypospadias repair is 6–12%, 88 and
Urethral breakdown hypospadias surgery is the major cause of urethral stric-
Treatment for urethral dehiscence will depend on ture formation in adults aged <45 years.89 Patients with
the length and extension of the neourethral defect, the strictures most commonly present with a weak stream,
amount of scar tissue encountered, and the availability with or without lower urinary tract symptoms. A dila-
of local skin.76 Thus, in order to select the appropriate tation of the urethra proximal to the stricture can be
repair option, the surgeon should check for persistent observed upon physical examination. Retrograde and
ventral curvature (and gauge whether this is secondary voiding cystourethrography (Figure 3) and cystoscopy
to contracture of the neourethra constructed during the are used to diagnose urethral strictures, as well as deter-
previous surgery) and assess the suitability of the residual mine the location, degree, and extension of the stricture.
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