Case Report
Rectourethral Fistulas: A Review
Amato A*
Department of Surgery, Hospital of Sanremo, Italy
*Corresponding author: Antonio Amato, Department of Surgery, Hospital of Sanremo, Via Borea 56–18038 Sanremo, Italy
Published: 15 Sep, 2016
Cite this article as: Amato A. Rectourethral Fistulas: A Review. Clin Surg. 2016; 1: 1121.
Abstract
Rectourethral fistula is an uncommon but challenging disease that may have a congenital or acquired origin. The most frequent etiologies are traumatic and iatrogenic. In the last decades the widespread use of radiation therapy for prostate cancer led to an increase of complex cases. The diagnostic work up is usually not demanding. Spontaneous closure can be achieved after single or double diversion in small, non irradiated fistulas. Many patients will ultimately require surgery and several surgical procedures have been described. The available literature shows retrospective studies of small series on single techniques and does not allow an objective comparison because patients and disease characteristics are markedly different among reports. The optimal therapeutic choice for every stage of the disease still remains controversial and no single procedure has been proven to be the best treatment.
Introduction
Rectourethral fistulas (RUF) in adults are a rare but potentially devastating condition requiring complex and demanding surgery. Most of the studies that have been published on this subject are retrospective, report small series and some of them are no ore than case-reports. Due to the rarity and complexity of this condition, epidemiologic data are lacking and no standardized algorithm exists to guide the surgeon in the management of these patients.
Classification and epidemiology
Culp & Calhoon et al. [1] described five groups of RUF according to their etiology: congenital due to malformation of the anus and the urinary tract; iatrogenic following surgery, radiotherapy or other treatment modalities; traumatic following fracture of the pelvis, gunshots or war wounds; neoplastic and inflammatory.
In a series of 23 male patients, Tiptaft et al. [2] found that the most common single causes of
RUF were fracture of the pelvis and iatrogenic causes (prostatic surgery, radiotherapy and urethral
instrumentation). Nyam et al. [3] reviewed a consecutive series of patients diagnosed in a 15 years
period. Out of 15 male patients treated for a prostatic cancer, 7 have had a radical retropubic
prostatectomy, 2 have had a radical retropubic prostatectomy after external beam radiation,
2 have had brachytherapy, and 3 were treated by a combination of external beam radiation and
brachytherapy. One patient showed a fistula after dilation of a urethral stricture. A systematic review
collected 416 patients from 26 papers [4]. A history of pelvic irradiation was found in 40% of the
cases, a figure that grew in the study period from 4% before 1997 to 50% after 1998. Combination
therapy was the most frequent radiation modality. In non-irradiated patients, etiology was iatrogenic
in 65%, traumatic in 22% and inflammatory in 6% of the cases respectively.
RUFs were also reported after prostate cryosurgery, high-intensity focused ultrasound therapy
(HIFU) and transrectal hyperthermia [5-8]. Barisic et al. [9] described 6 war wounds related RUF
in Bosnia-Serbia war, successfully treated by York Mason technique. Anecdotally a RUF was found
following rectal biopsy in irradiated patient or after sclerotherapy for piles [10-11]. RUF represents
one of the most devastating complications of prostate radiotherapy, graded as “IV” according to
the “Modified Radiation Therapy Oncology Group Lower Gastrointestinal Toxicity Scale” [12-13].
After prostate brachytherapy it occurred in 0.2–3 % of patients submitted to monotherapy, in 2.9
% treated by combined modality and in 8.8% who received salvage brachytherapy. Shah et al. [14]
found that the onset of symptoms occurs at a mean interval of 27.3 months after treatment.
After radical retropubic prostatectomy, most RUFs are due to neglected rectal injury occurring during dissection of the posterior prostate. This anatomic area is particularly critical for the surgeon because previous biopsies or episodes of prostatitis or locally advanced tumors increase the risk of adhesion. The prostatic urethra being adjacent to the rectal wall is the commonest site for fistulation. Less commonly, the bulbous and membranous urethra may be involved. If the wound can be directly visualized intraoperatively, a direct suture of the rectal wound is perfomed, avoiding systematic colostomy. Failure to notice the wound intraoperatively can lead to fistula formation. A review on 3834 radical prostatectomies found a
mean incidence of rectal injuries of 0.7% (range 0.2–2.9%), regardless of open or laparoscopic surgical approach [15]. Smith & Veenema et al. [16] reported their 20-year experience with 160 patients who had undergone radical retropubic prostatectomy with an incidence of 9.4% rectal injuries. In this group only 4 fistulas developed. Noldus et al. [17] reported 23 rectal injuries (3.9%) over 589 radical prostatectomies and cystoprostatectomy, with 12 RUF.
Case Presentation
Diagnosis
The diagnosis may be strongly suspected from the patient’s
history. The patients complain of the passage of gas or fecal particles
through the urethra or abnormal urethral discharge, passage of
urine through the rectum during micturition, recurrent urinary tract
infections. At the rectal examination a hard area in the anterior wall
of the low-middle rectum could be detected. The most important
diagnostic step is voiding cystourethrography that can demonstrate
the passage of the radiopaque contrast agent into the rectum through
the fistula tract. Proctoscopy and urethroscopy could be useful to
localize the enteric and urinary openings of the fistula. The distance
of the rectal opening from the anal verge is a significant parameter for
the subsequent therapeutic strategy.
Treatment
Especially in small fistulas, conservative treatment with urinary
catheter diversion and fully absorbable diet, alone or combined
with temporary colostomy, can achieve healing. Therefore, an initial
attempt with conservative treatment is reasonable. Diversion of urine
as well as correction of any stricture of the urethra distal to the fistula
and antibiotic therapy is the first-line therapy. In 1997, Noldus et al.
[17] reported that 7 out of 12 RUF after radical prostatectomy healed
spontaneously by long-term transurethral catheter.
While urinary diversion is mandatory, fecal diversion by means
of a temporary colostomy is controversial: it is mandatory as part
of a double diversion for some authors or selectively indicated
by others. It can prevent rectal wall distension and intraluminal
pressure, minimizing infection. Currently, colostomy could be
recommended when antibiotics alone cannot control fistula-
associated sepsis and severity of symptoms, when the fistula lies on
irradiated tissues, in recurrent fistulas, in large RUF (diameter >2 cm)
and in Immunosuppressed patients. A bridge-to-surgery diverting
colostomy with a 3-4 months interval before surgical treatment is
advocated in order to reduce the local inflammation. Hechenbleikner
et al. [4] reported that a double diversion was performed before
surgery for fistula in 89% of patients. In 1997, Al-Alì et al. [18]
reported 47% of spontaneous healing after double diversion alone
in a series of patients affected by war wounds, while healing after
diversion was noted by Chun in 60% of patients affected by RUF following laparoscopic radical prostatectomy [19]. On the other side,
Shah et al. [14] found that diversion of fecal stream does not heal RUF
after brachytherapy for prostate cancer.
Many patients will ultimately require surgery and it is often
technically demanding. Several approaches have been described
and more than 40 surgical techniques have been proposed with
variable rate of success [20-22]. However, no comparative study
exists; the published series are usually retrospective and show a
wide heterogeneity in terms of etiology, location, morphology of the
track and quality of the surrounding tissues. So, till now the optimal
operative management still remains controversial and there is no
consensus regarding the ideal treatment.
In 1958, Goodwin et al. [23] reported a series of 22 rectourethral
fistulas treated through a wide perineal approach. The procedure
requires an extensive mobilization of the rectum posteriorly and
the bladder anteriorly allowing the interposition of the levator ani
muscles between the urinary tract and rectum.
In 1885, Kraske described a posterior approach to the rectum that
required coccigectomy and partial sacrectomy, in order to perform a
rectal resection avoiding the challenge of a laparotomy. Some decades
later, Kilpatrick & Thompson proposed the same approach for the
treatment of RUF. After a fully circumferential mobilization of the
rectum proximally and distally to the track, the fistula was divided
sparing as much as possible on the urethral side. The rectal opening
was excised and closed, the urethra was repaired and stented with a
catheter.
In 1885, Kraske described a posterior approach to the rectum that
required coccigectomy and partial sacrectomy, in order to perform a
rectal resection avoiding the challenge of a laparotomy. Some decades
later, Kilpatrick & Thompson proposed the same approach for the
treatment of RUF. After a fully circumferential mobilization of the
rectum proximally and distally to the track, the fistula was divided
sparing as much as possible on the urethral side. The rectal opening
was excised and closed, the urethra was repaired and stented with a
catheter.
The York-Mason procedure [24] requires the division of the
rectal sphincters to give direct access to the RUF. It allows direct
visualization of the fistula via para-coccygeal trans-sphincteric
incision (from the tip of the coccyx to the anal canal). The patient
is placed in prone jackknife position with abduction of both lower
limbs and buttocks separated with adhesive tape. After incision and
sphincter division, the mucocutaneous junction and both internal
and external anal sphincter are marked by color-coded sutures to
provide a proper alignment and reconstruction at closure. The fistula
is excised exposing the catheter in the prostatic urethra and the rectal
wall is separated from the urinary tract by sharp dissection to allow a
sufficient mobilization. After closing the urethra, the rectum is sutured
paying attention that the suture lines do not overlap each other with a
“vest over pants” technique. Some authors suggest that suture should
be, if possible, covered with soft, healthy tissue, e.g. omentum, gracilis
muscle, scrotal flap, while others believe that it is not an essential part
of the procedure. The York-Mason procedure appears to be effective
and provides a superb exposure through unscarred tissue planes.
Published papers show it is a safe and reproducible technique with a
success rate of 91.7-100%, minimal morbidity and no impairment of
fecal continence [22,25-30]. Thus, for a long time it was considered as
first-line treatment of RUF after radical prostatectomy and has been
the most widely popular technique in the last decades.
Subsequently Parks et al. [31] described a fistula’s repair by
means of a full thickness flap of the anterior rectal wall through a
transanal approach, with the aim to avoid any division of the sphincter
mechanism. The rectal mucosa is excised laterally and distally to the
rectal opening of the track, denuding the circular muscular layer
of the rectum. Then a flap of about four centimeters in length is
performed. Dreznik et al. [32] described a similar procedure excising
the fistulous track to leave a transverse defect of 1–2 cm in the anterior
rectal wall. A longitudinal incision was made in the rectum from
each lateral edge of the defect for 3–4 cm proximally, performing a
U-shaped rectal flap. The width of the flap should be at least twice
than length. The defect in the urethra was closed using interrupted
absorbable sutures over the urethral catheter to prevent stenosis.
The rectal flap was advanced over the fistula and sutured to the
rectal wall with interrupted absorbable sutures ensuring the absence
of tension. Transanal flap repair showed a healing rate from 67 to
100% and the procedure is repeatable with the same effectiveness [32-34]. We proposed a sphincter-saving procedure through an anterior intersphincteric approach. The anterior rectal wall is sharply dissected
from the urethra until at least 2 cm above the fistula level. The track
is isolated and excised and the surrounding tissues are mobilized to
allow a tension-free repair. The urethral defect is closed using a single
row of 3-0 polyglactin interrupted sutures. Transanally a U-shaped
full-thickness rectal flap is advanced over the rectal opening and
sutured with a 3-0 monofilament interrupted sutures, being careful
that the two suture lines lie in different planes. Five patients were
successfully treated with no fistula recurrence or impaired continence
after a median follow up of 2 years [35].
Direct repair is best suited for surgically acquired fistulas that
are relatively small and surrounded by healthy tissue. In the last
decades the number of patients submitted to multimodalities
treatment for prostate cancer has risen and consensually the risk to
develop a RUF has increased [12,13]. As a consequence, surgeons
more frequently have to deal with more complex situation than in
the past. Radiation fistulas are often large and necrotic, with poor
tissue quality and impaired wound healing and they cannot be safely
repaired with simple division and closure. Tissue interposition is
almost always required and it can be accomplished using a dartos or
gracilis flap via a perineal incision. Furthermore, tissue interposition
can be combined with other technique to provide a more versatile
surgical armamentarium. Youssef et al. [36] successfully treated 12
male patients by means of a perineal subcutaneous dartos pedicled
flap procedure. RUFs were traumatic in 8 cases, or were developed
after prostatectomy in 4 cases. Through an inverted U-shaped
perineoscrotal incision a dartos flap was interposed as a vascularized
tissue flap between the repaired rectum and urethra. In the follow up
period ranging from 9 to 42 months no recurrence was registered.
The gracilis muscle transfer (unilateral or bilateral flap) can
be used universally regardless of patient age, gender or body
type, for closure of vesicovaginal, urethrovaginal and prostato-
cutaneous fistulas as well as RUFs. It can reliably be harvested, and
is rarely lost if the dominant proximal vascular pedicle is carefully
preserved and an adequate subcutaneous tunnel is created to avoid
flap compression. A small comparative study including five other
techniques demonstrated the clear-cut advantage of transperineal
gracilis muscle interposition flap in a heterogeneous group of patients
[3]. Besides the well-known technical aspects related to the proper
dissection and repositioning of the muscle, the peculiar key-points
of the procedure include: a perineal incision that allows a complete
dissection of the urethra from the rectum proximally and distally to
the track as well as a urethroplasty if a concomitant urethral stricture
is present; the fixation of the tip of the gracilis muscle to the apex
of the prostate and anterior rectal wall above the level of the closed
fistula, so that the bulk of the muscle rests between the sutured rectal
and urethral defects. In a retrospective study, Ghoniem et al. [37]
reported a 100% healing rate at a mean follow up of 28 months in
a series of 25 patients with complex RUFs, 15 of which underwent
radiotherapy. Vanni et al. [38] performed 74 transperineal repair
using gracilis muscle flap in 68 cases and other interposition flaps in
6 cases, with a 100% healing rate in 35 non irradiated patients vs. 84%
in 39 irradiated patients. The greater omentum is an option but this
procedure involves laparotomy and deep anterior pelvic dissection
and it may not be feasible in patients who have undergone previous
abdominal surgery.
High-tech mini-invasive procedures were also proposed in the
last years. Felipetto et al. [39] closed a prostatic-cutaneous fistula
which complicated a Pseudomonas prostatitis by means of human
fibrin sealant (Tissucol®). Etienney et al. [40] successfully treated a
recto-urethrocutaneous track in Crohn’s disease. The biological glue
passively occludes the fistula, promoting the process of endogenous
fibrin deposit and cicatrization by stimulating fibroblast proliferation
and migration, neovascularization and reepithelialization. Autologous
fibrin glue was used by Venkatesh et al. [41] in the treatment of a
group of patients including complex anorectal fistulas, rectovaginal
fistulas and urethro-vesico-rectal fistulas. A success rate of 60% was
reported without major complications but patients with urinary tract
fistulas and acquired immunodeficiency syndrome failed to respond.
Synthetic biological glue (Glubran®) was successfully injected to
close a prostato-perineal fistula complicating an abdomino-perineal
resection of the rectum and a neobladder-ileal fistula [42]. These
small experiences suggest that the therapeutic use of biological glue is
a minimally invasive technique, without any significant complication
and side effect. The procedure is easily repeatable and does not affect
further surgery suggesting a role as first-line therapy before venturing
on complex and high-risk surgical procedures. A combined approach
was suggested by Verriello et al. [43] who obliterated a RUF following
radical prostatectomy injecting a fibrin sealant before performing a
rectal mucosal flap to close the rectal opening.
In 2007, Rivera et al. [44] developed a RUF staging system
combining the fistula location and caliber with the presence or absence
of an irradiated field. They aimed to achieve a rational–though not
evidence-based - repair algorithm allowing the selection of the most
appropriate technique for each patient. Stages I and II include non-
irradiated patients with low or high RUF respectively. Transanal and
transperineal (trans-sphincteric) procedures with rectal flap seem to
be appropriate in these conditions. The latter techniques are suggested
also in stage III irradiated patients with small (diameter< 2 cm) RUF.
Large (stage IV) and complex (stage V) RUF in an irradiated field
should be managed with a muscle interposition flap procedure. In
stages from III to V a diverting colostomy is performed 4-6 weeks
before definitive surgery.
Conclusion
RUF is an uncommon but challenging disease, and several surgical procedure with different approaches have been described. Surgery is technically demanding with a cumulative healing rate of 87.5% and with an overall permanent fecal diversion rate of 10.6% (25% in irradiated vs. 3.8% in non-irradiated cases) [4]. Retrospective studies, often from single institutions, highlight the single repair techniques, but until now no comparative prospective trial has been carried out due to the rarity and heterogeneity of this condition. Pertinent literature shows poor quality data that makes difficult objective comparison, and no consensus on the best treatment option can be defined. Most of the therapeutic choice lies on personal surgeon preference and technical expertise.
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