Accepted Manuscript

Management of anorectal malformation without ligation of fistula: An approach

preventing posterior urethral diverticula
Vaibhav Pandey , Ajay N. Gangopadhyay , Dinesh K. Gupta , Shiv P. Sharma ,
Vijayendar Kumar
PII:

S1477-5131(14)00154-5

DOI:

10.1016/j.jpurol.2014.04.016

Reference:

JPUROL 1710

To appear in:

Journal of Pediatric Urology

Received Date: 25 November 2013

Accepted Date: 9 April 2014

Please cite this article as: Pandey V, Gangopadhyay AN, Gupta DK, Sharma SP, Kumar V,
Management of anorectal malformation without ligation of fistula: An approach preventing posterior
urethral diverticula, Journal of Pediatric Urology (2014), doi: 10.1016/j.jpurol.2014.04.016.
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Title: Management of Anorectal malformation without ligation of fistula: An approach preventing posterior
urethral diverticula
Key words: Anorectal malformation; Rectourethral fistula; Posterior urethral diverticula; Anorectoplasty

Authors: Vaibhav Pandey1*, AN Gangopadhyay2, DK Gupta3, SP Sharma4, Vijayendar Kumar5

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M.B.B.S, M.S, M.Ch., Senior Resident, Department of Paediatric Surgery, Institute of Medical Sciences,
Banaras Hindu University, Varanasi, U.P.
Email address - sunny.imsbhu@gmail.com
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M.B.B.S, M.S, M.Ch., Professor Department of Paediatric surgery, Institute of Medical Sciences, Banaras
Hindu University, Varanasi, U.P.
Email address gangulybhu@rediffmail.com
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M.B.B.S, M.S, M.Ch., Professor Department of Paediatric surgery, Institute of Medical Sciences, Banaras
Hindu University, Varanasi, U.P.
Email address hodps@rediffmail.com

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M.B.B.S, M.S, M.Ch., Professor Department of Paediatric surgery, Institute of Medical Sciences, Banaras
Hindu University, Varanasi, U.P.
Email address drspsharmabhu@rediffmail.com
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M.B.B.S, M.S, M.Ch., Associate Professor Department of Paediatric surgery, Institute of Medical Sciences,
Banaras Hindu University, Varanasi, U.P.
Email address - drvkbhu@gmail.com

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*Address for correspondence

Dr. Vaibhav Pandey
Senior Resident
Department of Paediatric Surgery
Institute of Medical Sciences

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Management of anorectal malformation without ligation of fistula: An approach
preventing posterior urethral diverticula
Vaibhav Pandey *, Ajay N. Gangopadhyay , Dinesh K. Gupta, Shiv P. Sharma , Vijayendar Kumar

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Department of Paediatric Surgery, Institute of Medical Sciences, Banaras Hindu University, Varanasi, UP,
India

University, Varanasi, UP, India

E-mail address: sunny.imsbhu@gmail.com (V. Pandey).

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* Corresponding author. Department of Paediatric Surgery, Institute of Medical Sciences, anaras Hindu

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Abstract Objective: The posterior urethral diverticulum is a common urologic

complication requiring reoperations in anorectal malformation cases (ARM). We
present a series of 24 cases of male ARM managed without ligation of fistula.
Material and methods: A prospective study was conducted between July 2010 and

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June 2012 including male neonates with ARM, where rectobladder neck and
rectoprostatic fistula were approached by the abdominal route. The fistulous tract was
dissected to the distal-most possible length and was excised flush with the urethra

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without its ligation. A per-urethral catheter was placed in situ. A record was made of

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any features of urinary leak and a micturating cystourethrogram was performed at the
1-year of follow-up.

Results: Twenty-four cases of ARM, 16 with rectobladder neck fistula and eight cases
with rectoprostatic fistula were included. Of these, 12 had single-stage primary
abdominoperineal pull-through and 10 were managed by primary posterior sagittal
anorectoplasty. Two cases with colostomy during the neonatal period were managed
by laparoscopic assisted anorectoplasty at 6 months. None of the cases had a urinary

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leak during the postoperative period. All had a normal micturating cystourethrogram
at 1 year.
Conclusions: The approach of dividing fistula without ligation may prevent posterior

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urethral diverticula, but larger long-term follow-up studies are needed.

KEYWORDS

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Anorectal malformation; Rectourethral fistula; Posterior urethral diverticula; Anorectoplasty

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Introduction

Single-stage repair for anorectal malformation (ARM) has shown results equivalent to
staged procedures [1,2]. Various urologic complications have been reported following
surgical management of ARM [2,3]. The posterior urethral diverticulum (PUD) is one

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urological complication that often requires reoperation in ARM cases when

symptomatic [4]. PUD occurs because of incomplete dissection of the distal-most
aspect of the rectum from the urethra and a diverticulum develops from a retained

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part of the rectourethral fistula which balloons out as more urine is sequestered in the
pouch-like structure [5,13]. We present a series of 24 cases of male ARM managed

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without ligation of fistula, resulting in decreased incidence of PUD.

Material and methods

A prospective study was conducted in the department of Paediatric surgery S.S.

hospital, B.H.U. Varanasi, between July 2010 and June 2012 after approval from the
institutional ethics committee. A total of 108 male patients with ARM were admitted
during the study period. All male neonates with ARM with rectobladder neck and

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rectoprostatic fistula approached through the abdominal route were included in the
study. Neonates who were lost to follow-up or died during the postoperative period
were excluded. Classification of cases was done on the basis of clinical examination

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and invertogram. An invertogram was obtained 24 hours after birth in all the patients.
A total of 24 cases were included in the study. Twenty-two patients underwent singlestage repair in the neonatal period. Two cases underwent laparoscopic assisted

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anorectoplasty (LAARP) following colostomy. The fistulous tract was dissected to the
distal-most possible length and was excised flush with the urethra (Figs 1 and 2). After

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this, the rectum was pulled through and the anoplasty was performed. A catheter was
placed in situ. The catheter was removed on the 14th postoperative day. A record was
made of postoperative recovery, including any features of urinary leak and other
complications. Regular follow-up of all patients was done. Ultrasonography of the

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abdomen to evaluate the urinary tract was performed at 6 months. A micturating

diverticula.

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Results

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cystourethrogram was performed at 1 year after surgery for evidence of any urethral

Out of 108 cases of male ARM admitted to our unit during the study period, 24 cases
were included in study. The mean age of presentation was 4.23 1.24 days (range 26
days). Twenty-two patients underwent single-stage repair during the neonatal period.
Two cases underwent laparoscopic assisted anorectoplasty (LAARP) following
colostomy. In cases with a single-stage procedure, 12 had primary abdominoperineal
pull through (APTT) and 10 were managed by abdomino-posterior sagittal

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anorectoplasty (PSARP). Two cases that underwent colostomy during the neonatal
period were managed by LAARP at 6 months of age. In 16 cases there was a
rectobladder neck fistula, and in eight cases there was a rectoprostatic fistula. The

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catheter was removed on the 14th postoperative day in 23 patients. None of these
patients had urinary complaints or evidence of urinary leak during the postoperative
period. One patient had accidental removal of the catheter on postoperative day 7;

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this patient also did not have any urinary complaints. No intraoperative and
anaesthesia complications were recorded. Feeding was started on days 45. All

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neonates passed meconium an average of 48 hours after surgery. All patients have
regular 3-monthly follow-up. Two patients had mild bilateral hydrouretronehrosis at 6
months on ultrasonography of the abdomen. A micturating cystourethrogram (MCUG)
was normal in both cases. None of patients had any urinary complaints at the 1-year

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follow-up. All cases had a normal upper and lower urinary tract on ultrasonography of
the abdomen and MCUG at the 1-year follow-up.

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Discussion

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The single-stage procedure for ARM during the neonatal period achieves the same
long-term outcome as the conventional three-staged repair [1,2]. Many children still
experience significant debilities from potentially avoidable complications with urologic
sequelae, despite significant advances in the surgical management of anorectal
malformations [3,4]. Various urologic complications have been reported, the most
common are recurrent rectourinary fistulas, bladder injuries, urethral injuries, and PUD
[3]. PUD is one of the most common urological complications requiring redo surgery

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[4,5]. Pea et al. [7] presented 20 cases of PUD developing in children with
rectourethral bulbar fistulas, all operated through a transabdominal approach. In
another series of 30 patients of PUD managed over a 22-year period, 80% of the

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patients had an abdominoperineal or sacroperineal pull-through procedure [11]. We

have been evaluating the structural development of our follow-up cases of ARM using
magnetic resonance imaging (MRI) of the pelvis, and 213 cases have undergone MRI of

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the pelvis over the past 5 years [15]: 8% (17 cases) of these have shown the presence
of PUD on MRI of the pelvis. In 15 of these cases, the fistula was ligated via the

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abdominal approach, and in two cases the fistula was ligated via the posterior sagittal
approach. Thus PUD more commonly occurs in cases where the fistula is ligated
through transabdominal approach [7]. So, in this prospective study, we included only
those cases where the fistula was approached through the abdominal route. In a series

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by Alam et al. [11] the median age of presentation of symptomatic patients with PUD
was 3 years (range 4 months to 6 years). In our series, 15 asymptomatic cases with

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PUD were detected at a median age of 4.56 1.68 years, of which five cases were less
than 1 year of age (with a mean age of 8.12 1.56 months) and another seven cases

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were below 3 years (with a mean age of 2.98 0.45 years). This, 80% of patients
presented under 3 years of age. Two patients presented with complaints of dribbling
of urine at 6 and 8 months of age (Figs 3 and 4). On MCU both showed the presence of
PUD with grade 45 bilateral vesicoureteric reflux with no evidence of posterior
urethral valve. In both the cases, preoperative catheterisation was not possible as the
catheter kept going into the diverticulum; the preoperative MRI showed diverticula to
be accessible through the abdominal route; excision was performed through the

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abdominal approach without ligation. In both the cases diverticula were located at
bladder neck. Follow-up at 6 months showed complete resolution of vesicoureteric
reflux, suggesting that PUD was causing some degree of bladder outflow obstruction

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and the reflux was indicative of this. The posterior sagittal or laparoscopic approaches
have been preferred for excision of diverticula as they provide better access and thus
have decreased the chances of recurrence [11]. In the current series eight out of 24

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patients are being followed up for 3 years or more. All have no complaints and have a

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good urinary stream.

The mechanism of these diverticula is not clear. The histology of resected

posterior urethral diverticula typically shows colon-type mucosa [14]. Currarino [13],
who reported the first case of PUD, and most other authors believe that the

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diverticulum develops from a retained part of the rectourethral fistula and balloons
out as more urine is sequestered in the pouch-like structure [11,13]. Suboptimal
amputation of the rectum owing to inability to reach the fistula site is a major

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contributory factor in the development of the diverticulum. The incidence of PUD

appeared to have stagnated or declined in recent years as more surgeons become

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proficient at the posterior sagittal approach to repairing ARMs [1]. In the vast majority
of male cases, the distal rectum lies below the peritoneal reflection. In cases where the
rectum lies below, with a long common wall between the rectum and urethra, surgical
dissection is technically more challenging via the transabdominal approach
(abdominoperineal or laparoscopy). It has been suggested that dissection should go on
till the urethra is reached and then the fistula should be ligated close to the urethra
[8]. The distal dissection to the urethra may damage it and both stricture and stenosis
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of the urethra are likely to occur. Thus a small diverticulum is usually left to avoid
dividing the fistula too close to the urethra and causing urethral stenosis and stricture
[9]. This tendency to err on the rectal side leads to division of fistula at some distance

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from the urethra, later growing into diverticula. After the division of a wide fistula or a
fistula opening lower down, the process of ligation further increases the length of
residual tissue and hence increases the chances of diverticula formation. Traction

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applied to the blind end of the rectum is required to demonstrate the fistula that
causes tenting of the urethra and increases the risk of transfixion of urethra with a

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stitch [10]. Chances of posterior urethral diverticulum are also high in laparoscopyassisted anorectal pull-through, especially when the technique is used to treat cases of
rectobulbar fistula [11]. A lower risk of injuries to the urethra has been reported
following simple division of the fistula without ligation [12]. Simple division of fistula in

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our series yielded good results in all cases. This also leads to a technically less
demanding procedure as one has to dissect the fistula to its most distal part and divide

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it without worrying about ligation. This reduces the chances of urethral injury. We
hypothesise that the distal stump of the fistula later sloughs and epithelisation occurs

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over the indwelling catheter. Long-term follow-up studies are needed to further
evaluate the advantages and disadvantages of this approach and occurrence of PUD in
these cases.
Conclusion
Rectobladder neck or rectoprostatic fistula in ARM cases approached through the
abdominal route can be managed by simple division of fistula without ligation. This

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approach may be useful in decreasing the incidence of PUD but a larger prospective
study with longer follow-up is required to establish the utility of this approach.

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Conflict of interests

None.

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Funding

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None.

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[15] Gangopadhyaya AN, Kumar V, Upadhyaya VD, Chowdhary S, Srivastava A,

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AQ1: The fistulous tract was dissected...: please check the edits to this sentence
AQ2: refs 3, 5, 6, 7, 12

AQ3: abdominoperineal pull through (APTT): please confirm that the abbreviation is
correct

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AQ4: On MCU both showed the presence of...:should MCU be MCUG here?
AQ5: the references should be mentioned in the text in the order they appear in the

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reference list. Please renumber and reorder. Also, there doesnt appear to be a text

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citation for reference 6. Please mention in the text or delete form the list

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Figure 1 Rectoprostatic fistula dissected as far below as possible.

Figure 2 Fistula divided without ligation.

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Figure 3 Magnetic resonance imaging showing posterior urethral diverticula.

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Figure 4 Micturating cystourethrogram showing the posterior urethral diverticulum and

bilateral dilating reflux.

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