Keywords classification - urethrocutaneous fistulas - reconstructive ladder
Introduction
Development of urethrocutaneous fistulas (UCFs) remains the main postoperative complication
in repaired hypospadias ranging from 12 to 90%.[1 ] Different modes of treatment for UCFs had been used by different authors, but none
of the studies had described such a classification that could directly point toward
the characteristics of UCFs, urethra, urethral plates, peno-prepucio-scrotal region,
and the associated morbidities like the hairy urethra, urethral stricture, urethral
diverticulum, urethral diverticulum with concretions, and the quality and quantity
of available dispensable peno-prepucio-scrotal tissues to provide a secured cover
to the closed fistulas, thus, this could have helped the operating surgeon to select
the best possible treatment option more quickly. In the present study, the treatment
ranged from the simplest conservative treatment (transurethral bladder decompression)
to the simplest surgical procedure by transection of the fistula tract (tractotomy),
purse-string closure, or multilayered closure (fistulorrhaphy). More complex surgical
repair required preputial or penile skin flaps (skin flap fistuloplasty), preputial
and scrotal dartos fascial waterproofing flaps, or the tunica vaginalis (TV) flaps
(waterproofing flap fistuloplasty) for reinforcement of closed fistulas. The most
complex procedures were closure of fistulas or redo urethroplasty and their re-enforcement
just like the Cecil-Culp procedure.[2 ]
Patients and Methods
This retrospective study comprised 68 UCFs of different categories (category A, n = 5; category B, n = 16; category C-a, n = 28; category C-b, n = 4; category D, n = 4; and category E, n = 11), developing into 68 repaired hypospadias in patients aged between 10 and 25
years, who reported for follow-ups in the “Hypospadias and VVFs Clinic” of Department
of Burns and Plastic Surgery, Postgraduate Institute of Medical Sciences, Rohtak,
between 2004 and 2016. Photographic records of (1) different types of UCFs, (2) different
characteristics of tissues of peno-prepucio-scrotal region (native and neourethra,
native and neourethral plate, and also the preputial hood, previous preputioplasty,
unused preputial hood, penile skin cover, corpora cavernosa, corpora spongiosum, quality
and quantity of scrotal skin, cryptorchism, unilateral undescended testis, inguinal
hernia, hydrocele, and varicocele), and thereafter (3) choosing for them the appropriate
treatment protocol constituted the material and methods for this study. Based upon
these three parameters, a clinical classification was designed ([Table 1 ]). The treatment plan was finalized after going through the details of (1) history,
(2) previous surgical interventions and its postoperative outcome, and (3) examination
of UCFs and tissues of peno-prepucio-scrotal region. This retrospective study was
performed (1) to categorize the UCFs and (2) to suggest appropriate treatment in a
pattern of a reconstructive ladder. This study was not aimed to know the incidence
and etiological factors of UCFs, that is, age of hypospadias, location of the original
meatus, type of single or staged original repair or the subsequent surgical repair,
and the isolated characteristics of UCFs like their location or size. This clinical
classification has incorporated all factors that are of clinical use to the operating
surgeon while repairing UCFs. Repaired hypospadias below 10 years of age either did
not report in the hypospadias clinic or would have gone to the pediatric surgery department.
Table 1
Clinical classification of urethrocutaneous fistulas (UCFs) in repaired hypospadias
Category and number of cases
Basis for categorization: (1) urethral calibration and methylene blue dye test, (ii)
artificial penile erection, (iii) examination of native and neourethras, (iv) inspection
of UCFs, urethral plate and the perifistular tissues, and (v) thorough study of peno-prepucio-scrotal
region were routinely done for categorization of UCFs
Characteristics of the given UCF/UCFs
Appropriate management following reconstructive ladder[a ]
A (n = 5)
Healthy urethra and neourethra with thick perifistular healthy tissue
Acute ultra-micro-UCF with its unepithelialized tract
Conservative treatment by bladder decompression
B (n = 16)
Healthy urethra and neourethra with thick perifistular healthy tissue
Eccentrically located ultra-micro-UCFs with epithelialized tracts
Centrically located ultra-micro, micro-, and macro-UCFs with epithelialized fistula/tracts
(mature fistulas)
Transection of fistulous tract (tractotomy)
Purse-string closure or multilayered closure (fistulorrhaphy)
C-a (n = 28)
C-b (n = 4)
Healthy urethra and neourethra with thick perifistular healthy tissue and presence
of preputial and/or penile skin in the coronal sulcus. Five UCFs were recurrent with
a history of one or more failed surgical attempts
Healthy urethra and urethral plates is fit for re-tubularization. Availability of
plenty of loose and dispensable penile skin to provide secured cover of soft tissue
and skin
Macro-UCF with availability of thick tissue all around or in its vicinity
Mega-UCFs and near-total disruption of neourethra. Healthy reusable urethral plates
Repair and re-enforcement with skin and/or water proofing flaps (fistuloplasty)
Re-tubularization of broken down neourethral plates and an eccentric closure using
the D.Smith technique[3 ]
D (n = 4)
Healthy native and neourethra and a healthy neourethral plate sufficient to reconstruct
neourethra but acute deficiency of dispensable peno-preputial skin not enough to provide
a secured skin and soft-tissue cover to the neourethra against re-fistulization (recurrences)
Multiple UCFs, mega-UCFs, and near-total disruption of neourethra except glans. These
are complex UCFs
Multiple fistulas were converted in to one larger fistula. Six months later, tubularization
of the urethral plate after dorsal median urethrotomy and a Cecil-Culp procedure for
skin and soft-tissue cover (skin and waterproofing flap)
E (n = 11)
Fistulas with (i) hairy urethra, (ii) urethral stricture, (iii) urethral diverticulum,
(iv) urethral metaplasia, (v) long segment of a narrow strictured urethral plate,
(vi) balanitis xerotica obliterans, (vii) residual significant chordee, and (viii)
short reconstructed neourethra
The characteristics of UCFs do not matter in the presence of too many other associated
morbidities of the urethra. These are complex UCFs
Each one of the urethral morbidities were corrected as described in the section of
patients and methods
F (n = 8)
Miscellaneous UCFs (UCFs that are congenital, iatrogenic, self-inflicted, infective,
decubitus in paraplegics, and in malignancy. Preputial fistula is not UCF in the true
sense
All other UCFs not related to hypospadias urethroplasty
The treatment will depend upon the etiopathogenesis and other characteristics of UCFs
a Reconstructive ladder: Treatment starts from the simplest procedure and thereafter
proceeds toward the most complex procedures, that is, category A → conservative treatment;
category B → tractotomy, purse-string suture, and fistulorrhaphy; category C-a and
C-b → fistuloplasty (skin flaps or waterproofing flaps with eccentric multilayered
closure); category D → Cecil-Culp technique; category E → redo urethroplasty cum Cecil-Culp
procedure; and Category F → depending upon the etiopathogenesis and the characteristics
of the fistula.
Categories A, B, C-a, and C-b are simple UCFs. Every part and tissues are normal except
for the presence of UCFs, and simple surgical procedures are required.
Categories D and E are complex fistulas. These fistulas have shortage of healthy tissues
and require complex surgical procedures.
All the five acute ultra-micro-UCFs in category A were put on conservative treatment
by uninterrupted bladder decompression using an 8-Fr infant feeding tube for a period
of 10 more days after detection of leakage of urine. Five of 16 fistulas of category
B were treated with transection of the fistula tracts (tractotomy) through skin incisions
in coronal sulcus (n = 2) and the lateral side of the penile skin (n = 3; [Fig. 1 ]), 3 were closed using purse-string suture of the dissected ostium of fistula by
6–0 Vicryl on round-bodied needle ([Fig. 2 ]), and the remaining 8 underwent a multilayered closure of the dissected perifistular
soft tissue (fistulorrhaphy; [Fig. 3 ]). Eight of 28 category C-a fistulas were closed and reenforced by preputial skin
flaps designed from the tissues of previously unused skin present on either side of
coronal sulcus, 5 were reenforced by penile skin flaps (skin flap fistuloplasty; [Fig. 4 ]), and 15 were provided preputial dartos flaps (n = 4; [Fig. 5 ]), scrotal dartos flaps (n = 3; [Fig. 6 ]), and TV flaps (n = 8; waterproofing flap fistuloplasty; [Fig. 7 ]). Five of 28 category C-a UCFs were recurrent fistulas with a history of one (n = 3) or more (n = 2) failed surgical attempts, and all responded favorably with flap closure (fistuloplasty).
Another four UCFs, two cases each of mega-UCFs ([Fig. 8 ]) and near-total disruption of neourethras ([Fig. 9 ]), were kept in category C-b, wherein re-tubularization of their neourethral plates
was done and the loose penile skin on either side of the re-tubularized neourethral
plates was mobilized to provide a multilayered eccentric closure like the D.Smith
technique[3 ] ([Fig. 10 ]).
Fig. 1 Division of the fistula tract (tractotomy) through lateral penile skin incision.
Unstitched wounds are left to heal spontaneously.
Fig. 2 Subcoronal urethrocutaneous fistula (UCF) excised. Purse-string suture inserted and
tightened after removal of the cannula. Final closure is done transversally (inset).
Fig. 3 Circum-excision of the fistula and its multilayered closure with 6–0 Vicryl (fistulorrhaphy).
Excised ring of the urethrocutaneous fistula (UCF; inset).
Fig. 4 Right-sided penile skin flap designed. Flap sutured eccentrically on the left to
prevent recurrences (inset).
Fig. 5 Right preputial flap sutured on the left. Probed urethrocutaneous fistula (UCF; upper
inset). Preputial dartos flap raised from the left skin (middle inset) and sutured
on the right (lower inset).
Fig. 6 Scrotal flap sutured on the right eccentrically. Urethrocutaneous fistula (UCF) and
flap (upper inset). Loose scrotal dartos (middle inset) sutured over the fistula (lower
inset).
Fig. 7 Scrotal and tunica vaginalis (TV) flaps. Proximal penile urethrocutaneous fistula
(UCF) and left testis (upper inset). TV flap raised (middle inset) and sutured over
closed UCF (lower inset).
Fig. 8 Mega-urethrocutaneous fistula (mega-UCF). Preputial hood and fistulorrhaphy (upper
inset). Peno-preputial skin brought laterally (middle inset) and sutured on left (lower
inset).
Fig. 9 Eccentric skin closure. (1) Near-total disrupted urethra. (2) Plate and urethrotomy.
(3) Epithelial strips. (4) Tubularization. (5) Penile dartos (6) sutured eccentrically
on the right.
Fig. 10 Right penile skin dartos flap sutured on the left. Left penile skin sutured on the
right (D. Smith technique) to prevent suture line superimposition (inset).
In four cases of category D fistula, two had multiple fistulas, one had mega-UCF,
and other one was of near-total disruption of neourethra ([Fig. 11 ]). The multiple fistulas were converted into one larger fistula before repair by
incising their intervening skin bridges. The urethral plates of multiple fistulas,
mega-fistula, and near-total disrupted neourethra were re-tubularized after 3 to 6
months and a secured cover was provided using the Cecil-Culp procedure ([Fig. 12 ]). In near-total disruption of urethroplasty, the intact part of the glans was incised
before proceeding for repair in the form of a complete redo urethroplasty, else there
would be risk of occurrence of UCF at the coronal sulcus. In 11 fistulas of category
E, hairy urethra was present in two, stricture of the distal urethra in two ([Fig. 13 ]), stricture of the penile urethra with a diverticulum in three ([Fig. 14 ]) of which one diverticulum had concretions, scarring, and residual chordee in one
([Fig. 15 ]), long segment of narrow urethral plate on fistulotomy in one ([Fig. 16 ]), balanitis xerotica obliterans (BXO) in one ([Fig. 17 ]), and one had short reconstructed neourethra. Accordingly, the hairy part of the
neourethra was excised in one and electro-depilated in the other. A strictured urethra
in both was laid open and grafted with inner preputial full-thickness skin graft (IPFTSG)
to reconstruct the neourethral plate. The diverticulum was trimmed equal to the caliber
of the urethra and the stricturotomy site was resurfaced by the trimmed lining of
the diverticulum and the fistulas were excised. The concretion-affected urethra in
one was excised and replaced with IPFTSG. Long, narrow, and strictured urethral plate
in one case was replaced by tubed full-thickness skin graft. Excision of the BXO-affected
part of the urethra and prepuce was done and replaced with the buccal mucosal graft
in one case. Excision of scarred tissues and correction of a chordee by dorsal tunica
albuginea plication (DTAP) was done in one case. The short urethra was transected
in one case for doing subsequent interposition urethroplasty, but this patient was
lost to follow-up. Tubularization of the neourethral plates was done after 3 months
when tissues had matured to become soft and supple. The miscellaneous category F as
detailed in [Table 1 ] was excluded from the present study.
Fig. 11 Near-total disrupted urethra. (1) Intact glans cut. (2) Urethral plate. (3) Median
urethrotomy. (4) Epithelial strips tubed. (5) Cecil-Culp done.
Fig. 12 Ventral penile shaft and neourethra sutured to midline incised scrotum (Cecil-Culp
procedure). Multiple fistulas made one before repair (inset).
Fig. 13 Distal urethral stricture with proximal urethrocutaneous fistula (UCF). Laid open
stricture and harvested inner preputial full-thickness skin graft (IPFTSG; upper inset).
IPFTSG re-surfacing stricturotomy site to construct urethral plate (lower inset).
Fig. 14 Distal stricture, proximal diverticulum, urethrocutaneous fistula (UCF), and skin
incision. (1) Wide diverticulum trimmed. Fistula excised, stricture incised, and grafted
with trimmed lining. (2) Plate tubed and skin closed eccentrically.
Fig. 15 Ventral chordee on artificial erection. (1) Dorsal midline incision for dorsal tunica
albuginea plication (DTAP). (2) Penile shaft straightened. (3) Final closure with
drain.
Fig. 16 Multiple urethrocutaneous fistulas (UCFs) and strictured plate. (1) Graft harvesting.
(2) Scarred tissue and diseased plate excised, graft tubed, and anastomosed with native
urethra. (3) Cecil-Culp procedure for cover.
Fig. 17 Balanitis xerotica obliterans (BXO) affected prepuce, urethra and fistula excised,
and raw area covered by a buccal graft to make a plate. (1) Buccal graft from the
right cheek. (2) Buccal graft.
Results
There was no recurrence of fistula in any of the patients in categories A, B, C-a,
and C-b. One patient in category D had urinary leakage, which was closed successfully
during the second stage of the Cecil-Culp procedure while separating the penile shaft
from the scrotal attachment ([Fig. 18 ]). Urethral calibrations in follow-ups revealed normal-diameter urethra. One patient
in category E had residual diverticulum and required further trimming of the diverticular
wall equivalent to the dimensions of the urethra. Recurrent fistulas had successful
closure after fistuloplasty. None of these patients had requested for any cosmetic
correction. Five patients had reported for preputial reconstruction and the same was
denied for want of adequate dispensable peno-preputial skin to reconstruct the prepuce.
All had good penile erection without residual chordee. Three patients had come for
getting fitness for marriage and were counseled accordingly to explain about their
surgical procedures and potency to the parents of their would-be wives.
Fig. 18 Urethrocutaneous fistula (UCF) after a Cecil-Culp procedure. (1) Penis with UCF.
(2) Circum-dissected fistula and median urethrotomy. (3) Multilayered closure of the
fistula and scrotum.
Discussion
Different authors have mentioned different characteristics of UCFs like the pediatric
fistulas[4 ]; adult fistulas[5 ]; number and size of fistulas[4 ]; size, location, number, and nature of tissue surrounding the fistulas[6 ]; recurrent fistulas[7 ]; and fistulas associated with stricture and diverticulum.[8 ] There is mention of different modes of treatment like conservative treatment,[9 ] purse-string closure,[10 ] multilayered closure,[11 ] simple closure, local skin flap closure or closure with waterproofing interposition
layer,[6 ] and re-enforcement of closed fistula by TV flap[7 ] and other soft-tissue re-enforcement interposition flaps (STRIFs)[12 ] or redo urethroplasty.[13 ] Further, the UCFs have been divided into groups I, II, and III based upon their
sizes, multiplicity, and the recurrent and persistent nature[4 ]
[14 ] or the hypospadias patients have been assigned different groups as per their adopted
treatment protocol.[5 ] Different characteristics of UCFs have also dictated the treatment options and surgical
outcome to some extent.[6 ]
A thorough literature search has revealed that UCFs developing after repair of male
hypospadias have not been assigned any well-established classification of wide clinical
use wherein due consideration had ever been given collectively to (1) all the different
characteristics of UCFs; (2) associated morbidities of native and neourethral plates;
(3) presence or absence of associated morbidities of native and neourethra like hairy
urethra, stricture urethra, urethral diverticulum, urethral metaplasia, extensive
perifistular scarring with secondary chordee, and the short reconstructed neourethra;
(4) availability and dispensability of penile, preputial, and scrotal skins; and (5)
the treatment plan opted for the given fistula. Little bit of literature is available
about female hypospadias because of its rarity.[15 ]
The presented clinical classification of the UCFs has multiple merits. (1) It categorizes
the UCFs into simple (categories A, B, C-a, and C-b) and complex (categories D and
E) fistulas. Simple fistulas are those in which the native urethra, neourethra, native
urethral plate, neourethral plates, and the peno-prepucio-scrotal region are all normal
and require simple treatment protocols like conservative treatment, tractotomy, purse-string
closure, fistulorrhaphy, or fistuloplasty. Complex fistulas are those which alert
the operating surgeon that either the diseased urethra would require correction or
re-do urethroplasty or a secured cover to the repaired fistula would be required by
doing Cecil-Culp procedure. (2) It incorporates almost all the characteristics of
fistulas (single or multiple fistulas; simple or complex fistulas; ultra-micro, micro,
macro, mega, near-total disruption of the urethra; acute or chronic fistulas; centric
or eccentric fistulas; primary or recurrent UCF) that have direct bearing on choosing
appropriate treatment planning. (3) It helps the treating surgeon to directly choose
the appropriate treatment. (4) It helps in maintaining a detailed record of the characteristics
of the fistulas at presentation, discharge, and follow-ups. (5) It will transfer detailed
information of the UCF to the next surgeon just by mentioning the category and detailed
diagnosis of the fistula while referring the post-hypospadias UCFs to a higher center
for repair of recurrences. (6) Further, adherence to this clinical classification
of UCFs will help in evaluating the results of different nonsurgical or surgical procedures
attempted for the treatment of UCFs having similar characteristics or category. (7)
Multicentric studies on the management of different categories of fistulas using different
surgical techniques as per the expertise of the surgical team will give better evaluation
of a particular surgical technique for a specific category of fistulas and accordingly
the surgeons would be able to alter their surgical planning in future to achieve better
results.
Formation of complex urethrocutaneous fistulas (CUCFs) needs to be prevented by adopting
the general principles of plastic surgery and repair of the fistula, because their
subsequent correction could be difficult and time-consuming.
Conclusion
The classification of UCFs is of clinical significance and utility in research. Treatment
planning should follow the principles of reconstructive ladder. Normal saline-induced
artificial erection must be done to exclude the presence of secondary chordee. Complex
fistulas respond better with a flap cover. An extragenital epithelium like the buccal
mucosa must be considered where IPFTSG was not available to replace an unideal urethral
plate. The Cecil-Culp procedure provides reinforcement of the fistulas by waterproofing
the dartos flap and skin cover. Dispensable peno-preputial skin must be utilized to
design the skin and soft-tissue flaps.
