Ventral Onlay Buccal Graft for Urethrorectal Fistula : Case Report

dr. Nanda Daniswara, SpU.1

dr. Johan Rinto Even Napitupulu 2

1
Division of Urology, Dr Kariadi General Hospital/Medical Faculty Diponegoro University, Semarang

Indonesia
2
Resident of General Surgery, Dr Kariadi General Hospital/Medical Faculty Diponegoro University, Semarang

Indonesia
 Ventral Onlay Buccal Graft for Urethrorectal Fistula : Case Report

dr. Nanda Daniswara, SpU.1

dr. Johan Rinto Even Napitupulu 2

1
Division of Urology, Dr Kariadi General Hospital/Medical Faculty Diponegoro University, Semarang

Indonesia
2
Resident of General Surgery, Dr Kariadi General Hospital/Medical Faculty Diponegoro University, Semarang

ABSTRACT

Background: Recto-urethral fistula (RUF) is a rare surgical condition. It is devastating

and represent an important problem for the patient. RUF is a challenge for the urologist

and colorectal surgeon whose take the case. There are many causes of RUF such as

iatrogenic, open prostatectomies, radiotherapy, brachytherapy, urethral instrumentation

etc.

Case Report: A 2-years-old boy came to emergency room dr. Kariadi Hospital by the

major problem was a urine catheter came out from the anus 3 days ago. The patient has

undergone PSARP operation 12 days ago in Malformation Anorectal case. Abdominal

examination explained a urinary catheter came out from the anus and there was also seen

an extravasation urine on anus.

Discussion: From the examination, we found vital stoma on left lower abdominal, some

faecal production, and urine extravasation in the anal. According to the algorithm, a

surgical exploration must be performed. By the time of exploration, there was a urethral

stricture from urethra pars posterior to pars bulbosa. Then, we performed grafting from

buccal mucosa and pasted to urethra with silicone urinary catheter Fr. 6. The graft

suturing with polyglicolic acid 6/0.

Conclusion: Patient with urethrorectal fistula is one of rare cases. There is a possibility

for recurrence. Perineal approach for repairing RUF, combined with buccal mucosa graft

interposition, is a fulfill simple technique for successful fistula closure, especially in

repeat surgery. Anamnesis, physical examination and imaging studies are important to

avoid misdiagnosis. Treatment should be done quickly and appropriately to decrease

chance of disability or death.

Keyword : Recto-urethral fistula, surgical repair, buccal mucosa

 Introduction

Rectourethral fistula is a connection between the lower urinary tract and the distal part

of the rectum. RUF is a rare conditions and can be classified as congenital or acquired1.

Treatment of RUF is still challenging due to the rarity and complexity of this condition. In

some small RUF, spontaneous closure can be expected with fecal and urinary diversion.

Because such an outcome is rare, surgical repair is definitely required for most RUF2.

The use of buccal mucous graft as donor tissue in urethroplastic reconstruction

consists of autologous transplantation of nonkeratinized oral mucosa to the urethra for use in

the repair of a variety of urological defects3. In the 18 studies that only assessed onlay graft

placement, the success rate was 79,2% (642 cases,p=0,28). Of the studies in which buccal

mucous graft was used as an onlay graft the graft was placed ventrally in 10 (325 total cases)

with a success rate of 87,7%.3

. This case report presents a 2-year-old boy with urethrorectal fistula with history of

malformasi anorectal

Case Presentation

A 2-year-old boy came to emergency room dr. Kariadi Hospital with his mother

because of the urine catheter came out from the anus and urinary drainage from the anus.

On April 2016 patient has been done PSARP in case of Malformation Anorectal

without fistula and then outpatient. After several days the fistula appears, patient underwent

fistula closure surgery for 2 times, but the fistula recurs again and urine drainage from anus.

The patient never had severe illness before, normal birth history and normal term of

gestational age, no congenital disease, no allergic history, and no history of routine

medication. He was a child of a sufficient socio-economic of the family.

 Physical examination revealed a 2-year-old boy, body weight 11,5 kg, height 135 cm,

general appearance looks moderately ill, active. His Glasgow Coma Scale was E4M6V5 =

15, with blood pressure 110/70 mmHg, pulse rate 90x/min (regular, volume and tone were

enough), respiration rate 20x/min (regular, deep of breath normal, no retraction), temperature

36,7oC. There was no anemic on both conjunctivas, isochoric pupil with diameter 3mm/3mm

and positive reflex pupil. Chest examination revealed symmetrical chest expansion, no injury

mark, normal breath sound without wheezing or rhonchi, normal heart sound, neither murmur

nor gallop. The abdomen looked normal with stoma on left lower region, the stoma looks

vital and there is stool production, no prolapse stoma. The abdomen was soft, no pain around

the abdomen, normal bowel sound. Penis and scrotum were normal. In the anal region urine

is seeping Motoric and sensory status in both lower limbs were normal, no cold acral and

Capillary Refill Time < 2”.

Laboratory findings showed Hemoglobin 10,8 gr%, Hematocrit 34,2%, Leucocyte

18.200 mmc, thrombocyte 606.000 mmc, blood glucose 77 mg/dL, Urea 27 mg/dL,

Creatinine 0,6mg/ dL, Sodium 133 mmol/mL, Potassium 3,2 mmol/ L, Chloride 95 mml/ dL,

PPT 11,4 (controlled : 11,3), APTT 39,1 (controlled : 31). Imaging studies were performed

including Bipolar Urethrosistografi.

 Bipolar Urethrosistografi

The patient was diagnosed with Fistula urethrorectal, stricture of posterior urethra,

malformasi anorectal on colostomy post PSARP. He was given with Dextrose 5% NaCl

0,25% 240 ml/24 hours intravenous, cefotaxim 250 mg/ 12 hours intravenous, and plan to

take operation procedures urethral reconstruction with buccal graft. Then after he had

surgical procedures, he had to stay in hospital for 5 days to be monitored his complaint, vital

sign, Glasgow Coma Scale, mobilisation,stoma production, and urine production from

cystostomy. If there was no complications happened, he could be discharge and schedule for

routine visits at urology department clinic.

 Discussion

Urethrorectal fistula is a rare but devastating disease that raises questions regarding

the most appropriate surgical approach. Most of the reported series claiming the superiority

of their technique are based on a small number of cases, and even in these, the ultimate

success was achieved after several attempts. Only few patients had successful repair after

first shot.4

The etiology varies, but can be classified as congenital, iatrogenic, traumatic,

neoplastic, and inflammatory.Asmuchas60%ofcasesarethoughttobeiatrogenic.4 Congenital

fistulas are rare and are most commonly associated with anorectal malformation disorders. In

these patients, fistulas either coexist with the malformation of the anus and rectum or can be

the results of the surgical correction of the malformation.5 Spontaneous closures of small

RUFs have been reported following long-term urethral catheterization. Spontaneous closures

following double diversion have also been reported in war wounds and post radical

prostatectomies. Currently, the widely accepted treatment protocol worldwide is double

diversion followed by definitive surgical repair.6

Iatrogenic RUF have a devastating impact on the patient’s quality of life and confront

the physician with a difficult challenge. Surgical repair is complex and the multiplicity of

approaches used for RUF are testimony to the methodical difficulties, often resulting in high

recurrence rates. The main problem in such repairs lies with the difficult access, via a

compound of tissues that have previously been operated on and even irradiated in some

cases.7

The diagnosis is made based on symptoms and appropriate diagnostic tests. The

primary symptoms are the presence of pneumaturia and/or faecaluria and the passage of urine

through the anus. The possibility of urinary infections is permanent. In variable proportions,
haematuria and perianal or perirectal pain may be added, occasionally a rectal examination

may allow the location or suspicion of the existence of the fistulous orifice. Diagnostic tests

are intended to confirm the presence of the rectourethral communication and its location and

to rule out the presence of a superimposed disease. The most often proposed studies are

rectoscopy, cystoscopy, urethroscopy and cystourethrography, with different authors

recommending them.8

In 1880, Duplay described a method for urethral construction in hypospadias that was

based on one of the basic principles in urethral reconstruction, consisting of the formation of

an epithelialized tube from a buried strip of skin. In 1949, Denis Browne described a similar

method for reconstruction of the urethra in hypospadias. Over time, the Duplay’s and Denis

Browne’s principle, according to which the buried strip of intact epithelium becomes an

epithelialized tube, has been widely used in reconstructive urology. In 1980, Monseur fully

applied Duplay’s principle and described the first dorsal urethroplasty. In 1996, Morey and

McAninch described the ventral onlay graft technique.9 Decisions regarding surgical

approaches for the treatment of complex urethral strictures associated with URF should be

based on a number of consideration including the location of the fistula, its aetiology, the

length of the urethral strictures, and a history of prior unsuccessful repair.10

BMG was first described for urethral reconstruction by Humby in 1941. It has become

an ideal urethral substitute because of ease of harvest, surgical handling characteristics,

hairlessness, and compatibility in a wet environment, its early in-growth and graft survival.

Because of these unique characteristics, buccal mucosa has endeared itself to the realm of

reconstructive urology. BMG also offer an inherent resistance to BXO. In the present series,

the substitution graft urethroplasty using buccal mucosa has a success rate of 95.35% at a

median follow-up of 58 month. There is controversy as to whether BMG should be placed

dorsally or ventrally. In the penile urethra, most experts would place it dorsally. In the bulbar
urethra, many experts place it ventrally, or mix ventral, dorsal and even lateral placement as

the clinical situation warrants. Multiple studies have shown that both dorsal and ventralonlay

BMG has good blood supply and mechanical support. The success rate for dorsal onlay is

reported between 85 and 100%.11

Table 1. Comparison Between dorsal vs. ventral BMG

Table 2. Complication of BMG

From the research conducted by Nikolaos Mertziotis et al, the use of buccal mucosa

graft and preputial graft as well in a single procedure to penile urethral strictures looks

provide adequate width of the urethra, and it can reduce the recurrence rate to minimum, even

in complicated cases.12 In 2008, Palminteri et al described the use of overlapping BMG for

reconstruction of bulbar strictures. At a mean follow-up of 22 months, an 89.6% success rate

was obtained in 48 patients with a mean stricture length of 3.65 c m.13

 BMG urethroplasty provides comparable medium-term results for reconstructing all

segments of the urethra, and appears to be the most versatile tissue available for

reconstruction, as it provides excellent results for both one- and two-stage urethroplasty.14

More recently Vanni et al published case series of 74 patients with rectourethral fistula which

included 2 patients with post- HIFU rectourethral fistula. There patients under went fistula

repair with interposition muscle flaps with or without BMG with overall success rate of

84%.15

For our case, after we performed anamnesis ang physical examination, we concluded

that this was fistulas urethrorectal with stricture of posterior urethra. Then we performed

urethral reconstruction with buccal graft, and debrided fibrotic tissue and then we take the

graft from the bucal mucosa, then we paste the graft to urethra and suture with poliglycolic

acid 6.0 and inserted a dower catheter.

The operation was successful and the patient was sent to pediatric ward. In pediatric

ward, he stayed about five days to monitor the complication, vital sign, Glasgow Coma Scale,
acute abdominal sign, monitor the operation wound ,and monitor the urine production both in

volume and color. The prognosis of this case was dubia ad bonam because this case was

treated accordingly and the urine doesn’t come out through the anus.

Conclusion

RUF being a rare condition does not have a wellestablished protocol for diagnosis and

treatment. However, It has been many reports by surgeons who have used different methods

of repair with varying degrees of success

The promising results we have achieved in repairing recurrent/persistent RUF using a

perineal approach and buccal mucosa interposition encourage us to continue with this

approach. Especially the ease of access to the RUF by using an approach familiar to most

urologists, and the simple procedure of harvesting the interpositional tissue compared with

other techniques.

Conflict of Interest

The authors declare that they have no conflicts of interests

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