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 Table of Contents  
CASE REPORT
Year : 2017  |  Volume : 7  |  Issue : 4  |  Page : 132-134

Post-caesarean vesicouterine fistula: Youssef's syndrome – A case report


1 Department of Obstetrics and Gynaecology, Kaduna State University, Kaduna State, Nigeria
2 Department of Radiology, Kaduna State University, Kaduna State, Nigeria

Date of Web Publication29-Oct-2018

Correspondence Address:
Dr. Matthew C Taingson
Department of Obstetrics and Gynaecology, Kaduna State University, Kaduna State
Nigeria
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ais.ais_40_17

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  Abstract 


Vesicouterine fistula (VUF) with cyclical hematuria, amenorrhea, and urinary continence is a rare condition referred to as Youssef's syndrome. It is most commonly observed after caesarean section (C/S) due to iatrogenic reasons. Open surgical management (transabdominal) has good results as it involves excision of the tract. We report a case of a VUF which developed after a repeat C/S and was successfully repaired with interposed vesico-peritoneal fold. Strict adherence to obstetric and surgical principles during Caesarean section can prevent formation of these fistulae.

Keywords: Caesarean section, vesicouterine fistula, Youssef's syndrome


How to cite this article:
Taingson MC, Adze JA, Bature SB, Amina DM, Caleb M, Amina A, Lydia A, Silas BT. Post-caesarean vesicouterine fistula: Youssef's syndrome – A case report. Arch Int Surg 2017;7:132-4

How to cite this URL:
Taingson MC, Adze JA, Bature SB, Amina DM, Caleb M, Amina A, Lydia A, Silas BT. Post-caesarean vesicouterine fistula: Youssef's syndrome – A case report. Arch Int Surg [serial online] 2017 [cited 2018 Nov 18];7:132-4. Available from: http://www.archintsurg.org/text.asp?2017/7/4/132/244413




  Introduction Top


Vesicouterine fistula (VUF) is an uncommon pathological communication developing between the uterus or cervix and the urinary bladder. VUF is a rare type of fistula and represents 1%–4% of all urogenital fistulas.[1] Caesarean section (C/S) accounts for 83%–93% of cases especially emergency C/S.[2],[3],[4] Youssef's syndrome classically presents as a triad of cyclical hematuria, amenorrhea, and urinary continence.[5] Other causes include obstetric injuries, for example, uterine rupture during vaginal birth after a caesarean delivery (VBAC) and use of obstetric forceps.[6] Less frequently, VUF can complicate inflammatory bowel diseases, endometriosis, intrauterine device migration, bladder tuberculosis, congenital lesions, placenta percreta, and manual removal of placenta.[7],[8] Newer cause includes uterine artery embolization.[9]


  Case Report Top


Mrs.SA, a 31-year old para 2, presented to the gynaecological clinic of Barau Dikko Teaching Hospital (BDTH) with a 2-year history of cyclical hematuria and secondary amenorrhea; there was no urinary incontinence. Symptoms dated back 3 years following her last child birth. She had a repeat emergency C/S due to poor progress in labor. Her first child birth, 6 years ago, was through a caesarean delivery for failed induction of labor. She was amenorrheic for a year after surgery, following which she developed cyclic hematuria with persistence of amenorrhea. She was a business woman married to a police officer, has no known drug allergy, does not consume alcoholic beverages, and is a nonsmoker.

Physical examination revealed an anxious young woman, afebrile with good hydration, and no pedal edema. Abdominal examination revealed a well-healed Pfannenstiel scar with no palpably enlarged organs. On pelvic examination, there was normal female external genitalia; speculum examination revealed a cervix that was 2 cm long, posterior smooth-surfaced. There was no urine leak from the anterior vaginal wall or from the cervix. Hematological and biochemical investigations were normal. Urine microscopy, culture and sensitivity, no organism was cultured after 48 hours. Ultrasonography of the abdomen and pelvis showed normal findings. Hysterosalpingography (HSG) showed immediate filling of the bladder through a fistulous tract, while the uterine cavity was not demonstrated [Figure 1]. She underwent a transabdominal transperitoneal repair. The uterovesical area was dissected through blunt and sharp dissection. The fistulous tract about 2 cm long between the pre-isthmic region on the uterus and the posterior wall of the bladder was exposed [Figure 2]a. The fistulous tract was excised; the defect on the posterior wall of the bladder was repaired in two layers with vicryl number 2/0 after the tissue was sufficiently mobilized. The defect on the uterus was closed in two layers as well [Figure 2]b. The bladder was drained with a urethral catheter. The postoperative period was uneventful, and urethral catheter was removed after 2 weeks. The patient made a full clinical and anatomic recovery; she had resumed regular menstrual cycles and had no urinary complaints. Postop HSG done was normal [Figure 3]. She was counseled on the need for booking in a tertiary facility in subsequent pregnancy as there is a small risk of recurrent fistula formation.
Figure 1: HSG depicting the fistulous tract

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Figure 2: (a and b) Excised tract showing the bladder and uterus

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Figure 3: Post repair HSG

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  Discussion Top


A classification of VUF based on the routes of menstrual flow has been proposed by Józwik and Józwik[10] which divides VUF into three types. Type I, characterized by the triad of amenorrhea, menouria, and complete continence of urine, is known as Youssef's syndrome. Type II is associated with dual menstrual flow through both the bladder and vagina. Type III is associated with normal vaginal menses and lack of menouria.[10] This patient qualified into type I VUF; the presence of a supraisthmic fistula helped in maintaining continence, leading to a delay in patient presenting to a gynecologist and its subsequent diagnosis. Hysterography and cystoscopy remain the “gold standard” in diagnosis.[11] This patient did not have a cystoscopy done as the facility was not available at our hospital at the time she presented. Transabdominal ultrasound evaluation of urogenital fistulae, supported poorly by the limited data available, has shown variable sensitivity in diagnosis of genitourinary fistulae, with some case reports reporting positive identification of fistula in as low as 29% of cases.[12] Ultrasound done in this case was essentially normal. The available data on magnetic resonance imaging (MRI) diagnosis of VUF are very limited.[12] In a study with a small number of patients by Abou-El-Ghar et al.,[13] 100% sensitivity was achieved in diagnosis of VUF. A few other authors have also reported accurate detection of VUF on MRI.[14] Conservative management with continuous bladder drainage for 4–8 weeks along with antibiotics and anticholinergics is recommended in cases where a small fistula is detected in the early or immediate postpartum or postoperative phase. The success rate of conservative management is less than 5%.[15] Transvesical fulguration with continuous catheterization and hormonal treatment can be successful for treatment of VUF with a small, well-epithelialized orifice, although the results in the presence of extensive scarring are poor and recurrence is high.[16] Open surgical management with transabdominal correction management has good results[15],[16],[17] as it involves excision of the tract. Transposing graft gives better results. In our case, we interposed vesico-peritoneal fold. The patient made a full clinical and anatomic recovery, confirmed by postop HSG [Figure 3]. Careful monitoring will be required in subsequent pregnancies as there is a small but potential risk of scar dehiscence and/or recurrent fistula formation. Youssef's syndrome, an uncommon if not rare entity, has been presented here. Meticulous adherence to obstetric and surgical principles during C/S and other obstetric interventions can prevent formation of these fistulae.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Lenkovsky Z, Pode D, Shapiro A, Caine M. Vesicouterine fistula: A rare complication of caesarean section. J Urol 1988;139:123-5.  Back to cited text no. 1
    
2.
Eisenkop SM, Richman R, Platt LD, Paul RH. Urinary tract injury during Caesarean section. Obst Gynecol 1982;60:591-6.  Back to cited text no. 2
    
3.
Buchholz NP, Daly-Grandeau E, Huber-Buccholz MM. Urological complications associated with caesarean section. Eur J Obstet Gynecol Reprod Biol 1994;56:161e163.  Back to cited text no. 3
    
4.
Yossepowitch O, Baniel J, Livne PM. Urological injuries during Cesarean section: Intraoperative diagnosis and management. J Urol 2004;172:196e199.  Back to cited text no. 4
    
5.
Youssef AF. “Menouria” following lower segment caesarean section; a syndrome. Am J Obstet Gynecol 1957;73:759-67.  Back to cited text no. 5
    
6.
Nouira Y, Feki W, Rhouma SB, Salah IB, Horchani A. Vesicouterine fistula as a complication of forceps delivery: A case report. Int Urogynecol J Pelvic Floor Dysfunct 2005;16:512-4.  Back to cited text no. 6
    
7.
Setubal A, Clode N, Bruno-Paira JL, Roncon I, Gracia IM. Vesicouterine fistula after manual removal of placenta in a woman with previous caesarean section. Eur J Obstet Gynecol Reprod Biol 1999;84:75-6.  Back to cited text no. 7
    
8.
Alkatib M, Franco AV, Fynes MM. Vesicouterine fistula following caesarean delivery – Ultrasound diagnosis and surgical management. Ultrasound Obstet Gynecol 2005;26:183-5.  Back to cited text no. 8
    
9.
Sultana CJ, Goldberg J, Aizenman L, Chon JK. Vesicouterine fistula after uterine artery embolization: A case report. Am J Obstet Gynecol 2002;187:1726-7.  Back to cited text no. 9
    
10.
Józwik M, Józwik M. Clinical classification of vesicouterine fistula. Int J Gynecol Obst 2000;70:353-7.  Back to cited text no. 10
    
11.
Bhattacharjee S, Kohli UA, Sood A, Tripathy S, Gupta M. Vesicouterine fistula: Youssef's syndrome. Med J Armed Forces India 2015;71:s175-7.  Back to cited text no. 11
    
12.
Adetiloye VA, Dare FO. Obstetric fistula: Evaluation with ultrasonography. J Ultrasound Med 2000;19:243-9.  Back to cited text no. 12
    
13.
Abou-El-Ghar ME, El-Assmy AM, Refaie HF, El-Diasty TA. Radiological diagnosis of vesicouterine fistula: Role of magnetic resonance imaging. J Magn Reson Imaging 2012;36:438-42.  Back to cited text no. 13
    
14.
Murphy JM, Lee G, Sharma SD, Doble A, Lomas DJ. Vesico-uterine fistula: MRI diagnosis. Eur Radiol 1999;9:1876-8.  Back to cited text no. 14
    
15.
Porcaro AB, Zicari M, Zecchini Antoniolli S, Pianon R, Monaco C, Migliorini F, et al. Vesicouterine fistulas following cesarean section: Report on a case, review and update of the literature. Int Urol Nephrol 2002;34:335-44.  Back to cited text no. 15
    
16.
Hadzi-Djokic JB, Pejcic TP, Colovic VC. Vesico-uterine fistula: Report of 14 cases. BJU Int 2007;100:1361-3.  Back to cited text no. 16
    
17.
Drissi M, Karmouni T, Tazi K, EL Khader K, Koutani A, Ibn Attya A, et al. Vesico-uterine fistulas: An experience of 17 years. Prog Urol 2008;18:173-6.  Back to cited text no. 17
    


    Figures

  [Figure 1], [Figure 2], [Figure 3]



 

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