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106
List of Recommendations
1. Evaluate patients with complex perianal Crohn’s disease with MRI or EAUS in
conjunction with examination under anesthesia to assess proximal disease and
anatomy of the perianal disease (strong recommendation based on moderate
quality evidence).
2. For patients with complex perianal Crohn’s disease complicated by sepsis, the
initial step is to drain all abscesses and place loose setons in identiable stulas
(strong recommendation based on low quality evidence).
3. Patients with refractory perianal symptoms and septic complications requiring
recurrent surgical intervention, and patients with signicantly impacted quality
of life despite combined seton and medical management should be counseled to
consider fecal diversion with a loop ileostomy (strong recommendation based on
low quality evidence).
4. Counseling regarding temporary fecal diversion should include discussion about
a possible permanent ostomy given low rates of continuity restoration (moderate
recommendation based on moderate quality evidence).
5. Discussion about permanent diversion is especially pertinent for patients with
proximal disease who have or are at risk for rectal stenosis (strong recommenda-
tion based on moderate quality evidence).
Personal View oftheData
In our search, no studies directly compared seton placement with diversion in
similar patient populations. Clinically, these surgical strategies are used in a step-
wise approach. Drainage of any infectious source and seton placement are the rst
stage of perianal Crohn’s disease treatment. This is completed in conjunction with
medical treatment, specically anti-TNF biologic therapy. If seton placement
with concurrent maximized medical management fails, then diversion is consid-
ered. The discussion of diversion is based on the severity of complications, includ-
ing the frequency of surgical interventions for perianal Crohn’s disease, fecal
incontinence, patient discomfort, and the combined effect of these complications
on the patient’s quality of life. Proceeding with diversion is a joint decision
between patient, surgeon, and the gastroenterologist. The decision is based on
exhaustion of combined seton and medical therapy, and the potential for improv-
ing quality of life [77].
Diversion with a loop ileostomy is generally discussed as an initial temporizing
measure that allows treatment of perianal disease with biologic agents in the absence
of continued irritation from the fecal stream. However, temporary diversion has
been shown to be reversed in as few as 17% of patients [39, 43]. Therefore, patients
should be made aware that a temporary loop ileostomy may result in permanent
diversion with or without conversion to proctectomy.
S. Khaitov and A. Ofshteyn
107

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11 Management ofSevere Anoperineal Disease
113
© Springer Nature Switzerland AG 2019
N. Hyman et al. (eds.), Mastery of IBD Surgery,
https://doi.org/10.1007/978-3-030-16755-4_12
C. Twum-Barima, BSc (Hons), MBChB, MRCS · P. Tozer, BSc (Hons), MBChB, MRCS (*)
Robin Phillips Fistula Research Unit, St Mark’s Hospital, London and Imperial College
London, London, UK
e-mail: philtozer@nhs.net
12
Management ofAno/Rectovaginal
Fistula
CharleneTwum-Barima andPhilTozer

Introduction

Fistulas to the vagina from the anus or low rectum are variously called anovaginal
and rectovaginal stula (RVF) and the latter phrase will be used throughout this
chapter. RVF’s are a devastating problem producing severe symptoms in many
cases. They occur more commonly in Crohn’s disease than in the general popula-
tion, with a prevalence of 10% and mean age of onset of 34years in a series of 886
patients [1]. A population based survey from Minnesota found that 35% of Crohn’s
disease patients had stulas and 9% of these were RVF’s [2]. RVF’s are more com-
monly associated with colonic (23%) than small bowel (3.5%) Crohn’s disease [3,
4]. A variety of techniques are used in managing Crohn’s disease related RVF’s. In
this chapter, the literature pertaining to surgical treatment of an RVF complicating
Crohn’s disease is reviewed and is followed by recommendations and opinions
(Table12.1).
Table 12.1 PICO question
PICO table
Population Intervention Comparator
Outcomes
Management of ano/
rectovaginal stula
Pts with Crohn’s
disease
repair stoma Recurrent stula,
quality of life
114

Search Strategy

A comprehensive literature search of the Cochrane, EMBASE and MEDLINE data-
bases was performed to identify all relevant English-language publications over the
last 20years. Key search terms included: ‘rectovaginal stula,’ ‘anovaginal stula,’
‘Crohn’s’ and ‘surgery’ (and variants). Studies were excluded if they reported fewer
than 3 patients with Crohn’s RVF, or did not study surgical intervention (reparative
or ostomy formation), describe outcomes in Crohn’s patients separately from other
etiologies or consider stula healing and/or quality of life assessment as outcomes.
Conference proceedings were excluded. Only the most recent study was included if
similar studies from the same institution were encountered. The references of the
included studies were reviewed to identify additional studies.

Results

Almost all published studies are observational and retrospective in nature. No stud-
ies directly compare reparative surgery with ostomy formation. Reparative surgical
options reported include gracilis interposition, advancement ap repair (i.e. rectal,
vaginal), Martius ap, episioproctotomy, abdominal approaches including Soave-
type coloanal pull-through, and novel approaches including glue, plug, mesh inter-
position, stem cell injection and fat injection. Ostomy formation was performed in
three situations: temporarily to “protect” a repair; permanently to reduce symptoms
by defunctioning the rectum and therefore the stula, or; as part of an ablative oper-
ation (e.g. proctectomy).
Studies either tend to be of a single technique with multiple etiologies, or mul-
tiple techniques with a single etiology, or multiple techniques in multiple etiologies.
Many studies had only a small number of Crohn’s disease RVF’s treated by a par-
ticular technique and comparison between techniques is therefore difcult. It was
not always possible to determine the precise number of Crohn’s disease RVF
patients undergoing a particular technique. Most studies measured healing of the
stula as an outcome although assessment was performed at various time points
using multiple methods. Quality of life scoring was rarely reported. Heterogeneity
in etiology, stula morphology, surgical technique, previous repair attempts, and
outcome measurement limits the strength of the recommendations that follow
(Table12.2).
Few series of gracilis interposition contain three or more Crohn’s disease RVF
patients. Most include recurrent stulas and utilize fecal diversion. In one study,
three of nine patients’ RVF healed and one became sufciently small with reduced
symptoms such that no further treatment was required [5]; these four patients had
their stomas successfully reversed. Quality of life assessments were not reported. In
a study of gracilis interposition for RVF’s and rectourethral stulas, repair of
Crohn’s disease-related stulas was reported as “successful” but only one of the
three RVF’s healed [7]. Another study reporting 11 patients with Crohn’s disease
RVF’s treated with gracilis interposition found that all had healed and had their
C. Twum-Barima and P. Tozer
115
stoma reversed, although the follow-up interval was just a few months in one-half of
the patients [6] and medical treatment was not described.
One study of gracilis interposition for multiple etiologies reported quality of life
outcomes in addition to healing, which occurred in 4 of 5 (80%) Crohn’s disease
patients [8]. Quality of life was reported for all 8 RVF’s. Seven of eight women had
been sexually active before surgery but only four remained so afterwards. All four
had a healed stula. One noted reduced libido and two complained of dyspareunia,
but three of the four said they were satised with their sex-related quality of life.
Studies reporting outcomes from advancement ap repair also suffer from het-
erogeneity. For example, one study with 6 Crohn’s disease RVF patients used 5
vaginal advancement aps and 1 rectal advancement ap [11]. In the vaginal ap
patients, one was bolstered by porcine small intestinal submucosa-derived mesh,
one by gracilis interposition, and three by Martius aps, with only one patient hav-
ing follow-up longer than 4months. It is difcult in this context to dene advance-
ment ap success rates. Several studies include Crohn’s disease RVF’s but do not
report their outcomes separately from the RVF’s of other etiologies or anorectal
Table 12.2 Studies evaluating reparative techniques for Crohn’s disease RVF
Study
Patients Intervention(s) Outcome(s)
Quality
of evidence
Wexner (2008)
[5]
9 CD
RVF
Gracilis 3/9 (33%)
healed
Low
Furst (2008) [6] 11 CD
RVF
Gracilis 11/11 healed Low
Ulrich (2009)
[7]
3 CD
RVF
Gracilis 1/3 (33%)
healed
Low
Lefevre (2009)
[8]
5 CD
RVF
Gracilis 4/5 (80%)
healed
Low
Rottoli (2018)
[9]
8 CD
RVF
Gracilis 6/8 (75%)
healed
Low
Jarrar (2011)
[10]
12 CD
RVF
Rectal adv ap 5/12 (42%)
healed
Low quality
Nosti (2013)
[11]
6 CD
RVF
5 Vaginal adv ap, 1 rectal adv
ap, (4+Martius/Gracilis)
4/6 (67%)
healed
Very low
Songne (2007)
[12]
7 CD
RVF
Martius ap 8/8 (100%)
healed
Very low
Pitel (2011) [13] 8 CD
RVF
Martius ap 4/8 (50%)
healed
Low
Schwandner
(2009) [14]
9 CD
RVF
Rectal adv ap & Surgisis
mesh
7/9 (78%)
healed
Low
Mege (2016)
[15]
4 CD
RVF
Biological mesh (Strattice) 2/4 (50%)
healed
Low
Gajsek (2011)
[16]
8 CD
RVF
Button plug 4/8 (50%)
healed
Low
Garcia-Arranz
(2016) [17]
5 CD
RVF
Allogeneic stem cell transplant 3/5 (60%)
healed
Very low
Norderval
(2018) [18]
7 CD
RVF
Autologous fat graft injection 6/7 (86%)
healed
Low
12 Management ofAno/Rectovaginal Fistula
116
stulas to the perineum. One study that carefully described 12 Crohn’s disease
RVF’s found a primary healing rate of RVF managed by rectal advancement ap of
42% [10], rising to 83% after up to 3 attempts. Across the entire cohort, continence
and quality of life were similar before and after surgery. However, the “inconti-
nence” associated with the stula and true anal incontinence associated with the
surgical insult to the internal anal sphincter are not disentangled in this study and so
the true impact of this surgical technique on continence and any related impact on
quality of life cannot be determined.
Two studies analyzed the Martius ap technique in a variety of patients. One
study reported 8 Crohn’s disease RVF patients and found that the stula healed in
one-half [13]. Fecal incontinence (Wexner score), sexual quality of life [Female
Sexual Function Index (FSFI)], and overall quality of life [Short Form-12 (SF-12)
Health Survey] were analyzed for the entire group but results in the Crohn’s disease
RVF group were not separately presented. Fecal diversion was also used in several
patients and was maintained in at least one-half of the Crohn’s disease patients. An
earlier study described healing of all (7/7) Crohn’s disease RVF’s, but two of the
seven patients went on to proctectomy for “non-RVF” indications and the details are
unclear [12].
The generally poor outcomes associated with RVF repair, often reported as
worse in patients with Crohn’s disease, coupled with the morbidity of the operations
and postoperative symptoms, have prompted a slew of novel techniques, usually
aimed to be less invasive but more effective.
The over the scope clip (OTSC) has been used in Crohn’s disease RVF’s in two
studies of mixed stulas and etiologies. In one, [19] the success rate in the Crohn’s
disease RVF group was not described and in the other [20], only two Crohn’s dis-
ease RVF patients were included and neither healed.
Two studies reported biological mesh interposition in at least 3 Crohn’s disease
RVF patients. Porcine skin-derived mesh was used in 4 diverted patients in one
study [15]. Two patients’ RVF’s fully healed, one failed to heal, and the other was
converted to a rectoperineal stula. In another study, porcine small intestinal
submucosa- derived mesh was used and 7 of 9 (78%) Crohn’s disease RVF patients’
stulas were healed at the end of follow up [14].
A submucosal tissue-derived stula plug specically designed for stulas to the
vagina was used in 8 Crohn’s disease RVF’s without fecal diversion [16]. At nal
follow-up, 4 of 8 (50%) stulas were healed. Plug dislodgement led to several fail-
ures, and repairs in smokers and repeat procedures were always unsuccessful.
Allogeneic stem cells have been used to treat anal stulas arising in Crohn’s
disease in a large multicenter randomized controlled trial [21]. A phase I–II study
[17] from the same Spanish group found that 3 of 5 (60%) Crohn’s disease RVF
patients maintained a healed stula at 1-year follow-up. Autologous fat implanta-
tion led to healing of Crohn’s disease RVF’s in one study, in which 6 of 7 patients
(86%) had no stula at nal follow-up [18]. Repeat procedures, fecal diversion, and
medical treatment occurred but were not specied.
Several mixed series have been published that include Crohn’s disease RVF’s
treated with several different surgical techniques. In many it is impossible to
C. Twum-Barima and P. Tozer