|Year : 2018 | Volume
| Issue : 1 | Page : 101-104
Vesicouterine fistula (Youssef's Syndrome): A case report and review of literature
Ibrahim Garba, Ayyuba Rabiu, Zainab D Ahmed, Muhammad S Ozegya
Department of Obstetrics and Gynaecology, Bayero University Kano/Aminu Kano Teaching Hospital, Kano, Nigeria
|Date of Web Publication||23-Mar-2018|
Dr. Ayyuba Rabiu
Department of Obstetrics and Gynaecology, Bayero University Kano/Aminu Kano Teaching Hospital, Kano
Source of Support: None, Conflict of Interest: None
Vesicouterine fistula is a rare urogynecological fistula, usually iatrogenic following lower uterine segment cesarean section. The clinical features of cyclical hematuria, amenorrhea, and urinary continence, Youssef's syndrome, were first reported in the literature in 1957, among patients with vesicouterine fistula. The case reported was a 36-year-old multipara who had two previous lower segment cesarean sections and presented with amenorrhea and menouria. Hysterosalpingography confirmed the diagnosis. The fistula was repaired following the transabdominal surgery. She regained normal vaginal menses.
Keywords: Cesarean section, hysterosalpingography, transabdominal repair, vesicouterine fistula, Youssef's syndrome
|How to cite this article:|
Garba I, Rabiu A, Ahmed ZD, Ozegya MS. Vesicouterine fistula (Youssef's Syndrome): A case report and review of literature. Niger J Basic Clin Sci 2018;15:101-4
|How to cite this URL:|
Garba I, Rabiu A, Ahmed ZD, Ozegya MS. Vesicouterine fistula (Youssef's Syndrome): A case report and review of literature. Niger J Basic Clin Sci [serial online] 2018 [cited 2021 Jun 23];15:101-4. Available from: https://www.njbcs.net/text.asp?2018/15/1/101/228369
| Introduction|| |
A vesicouterine fistula (VUF) is a pathological communication between the epithelial surfaces of the bladder and the uterus or cervix.,,, It is a rare form of urogynecological fistula when compared with vesicovaginal fistula and represents about 1–4% of all cases of urogynecological fistula. It is usually iatrogenic following cesarean section in about 83–93% of cases.,, Other least common causes of vesicouterine fistula include induced abortion, obstructed labor, dilatation and curettage, vaginal birth after cesarean section, placenta percreta, forceps delivery, brachytherapy and in rare occasions, migrated intra uterine contraceptive devices.
Patients with vesicouterine fistula classically present with cyclic hematuria (menouria), absence of vaginal bleeding (amenorrhea), and complete urinary continence. In rare occasions, urinary incontinence could also be a clinical presentation in patients with vesicouterine fistula, especially with the involvement of the cervix or the fistula is infra isthmic.
Vesicouterine fistula was first reported in the literature in 1908 by Knipe. However, it was described by Youssef in 1957 as a clinical syndrome consisting of cyclic hematuria (menouria) amenorrhea, and complete urinary incontinence in a patient who had lower segment cesarean section. Youssef was also the first to coin the term “Menouria” resulting from VUF. A suggested classification of VUF, based on the routes of menstrual flow proposed by Józwik and Józwik  in 2000, divides vesicouterine fistula (VUF) into three types. Type I is characterized by the triad of amenorrhea and menouria, and the complete continence of urine has been known as Youssef's syndrome; Type II is associated with dual menstrual flow via both the bladder and vagina, and Type III is associated with normal vaginal menses and lack of menouria.
| Case Report|| |
She was a 36-year-old multipara whose last normal menstrual period was 8 years prior to presentation, before her last child birth. She presented with a 7 year history of amenorrhea and cyclical passage of bloody urine. Her problems started 8 years ago when she had a repeat emergency cesarean section, following prolonged labor and failed vaginal birth after cesarean section at a private facility. Following the surgery she had urethral catheter in situ for 37 days, which was draining bloody urine. There was no drainage of lochia per vaginum, and she was oblivious of suspected bladder injury. She continued to breastfeed and did not menstruate for about a year. Thereafter, cyclical hematuria was noticed, which lasted 4 days every month. No cyclical bleeding per vaginum, and she was continent of urine. She was seen in the same hospital on account of dysuria and lower abdominal pain. Ultrasound scan revealed normal findings and urine microscopy, culture, and sensitivity cultured E. coli, which warranted treatment for urinary tract infections. She, however, continued to experience cyclical hematuria, amenorrhea, and cyclical mild pain on passing urine, which necessitated referral to our hospital.
She attained menarche at the age of 15 and menstruated for 5 days in a regular cycle of 30 days. Her first pregnancy was in 2006; she had emergency cesarean section on account of the prolonged labor at the referring hospital. She delivered a live female baby, who cried immediately after birth and was fully immunized for age. Her second pregnancy was in 2009, she had emergency cesarean section on account of the prolonged labor (failed vaginal birth after cesarean section) at the same health facility. The baby cried immediately after birth and was fully immunized for age. She was transfused 2 units of blood because of postpartum hemorrhage.
She was a known hypertensive, diagnosed a year ago, and regular on antihypertensives. She has no drug allergy. She was a seamstress, married in a monogamous setting to a civil servant. She neither smoke cigarettes nor ingest alcoholic beverages.
On examination, she was a young woman, afebrile, not pale, not dehydrated, and not jaundiced. Her pulse rate was 80 beats/min and blood pressure was 130/70 mmHg. Abdominal examination revealed a full abdomen that moved with respiration, with a midline sub-umbilical scar. There were no areas of tenderness, liver and spleen were not palpably enlarged, and the kidneys were not ballotable. Vaginal examination revealed a normal female external genitalia, healthy looking cervix with no adnexal masses and no leakage of urine.
An assessment of utero-vesical fistula was done following hysterosalpingography under fluoroscopic guidance, which showed continuous opacification of the urinary bladder through the uterine cavity [Figure 1], [Figure 2], [Figure 3], [Figure 4].
|Figure 2: An HSG depicting the pelvic region with leech wilkinson cannula in situ|
Click here to view
|Figure 3: An antero-posterior view of the pelvis showing continuous opacification of the urinary bladder through the uterine cavity|
Click here to view
|Figure 4: A lateral view of the pelvis showing continuous opacification of the urinary bladder through the uterine cavity|
Click here to view
The findings and diagnosis were discussed with her. A client media-released consent and consent for the surgery were sought and obtained.
She had exploratory laparotomy and repair of the fistula under general anesthesia. A 16G Foley catheter was passed before the exploration. A fistulous tract was noted between the dome of the bladder and the isthmic region of the uterus about 4 cm in length. The tract was adherent to the uterus at a point corresponding to the site of the lower uterine segment incision of the antecedent cesarean section. The fistulous tract was carefully dissected off the uterus, and the presumed lower and upper lips of the transverse lower segment incision were held with Allices. The bladder was closed in two layers with Vicryl 2/0 using an “inverting suture technique.” The uterine defect was closed in single layer with vicryl 1. Hemostasis was secured, and the rectus sheath and the skin were closed with vicryl 1 and nylon 2/0, respectively. The Foley catheter was kept in situ for 14 days. She was commenced on intravenous antibiotics, fluids, and analgesics for the first 24 h of surgery. Thereafter, antibiotics were converted to oral for 5 days and were since commenced on oral fluids of 4–6 liters per day for 2 weeks. Urine sample was collected for microscopy, culture, and sensitivity on days 3, 5, 7, and 10, and the catheter tip was cut and sent for microscopy culture and sensitivity on day 14, which revealed no growth. A follow-up visit after 4 weeks of discharge revealed remarkable findings. She observed normal menstrual flow for 4 days. There was no cyclical hematuria.
She was counseled for contraceptives for at least 2 years before her next pregnancy and planned cesarean section subsequently.
| Discussion|| |
Vesicouterine fistulas have been reported as the least common urogynecological fistulas. Typically, patients present with cyclic hematuria (menouria), amenorrhea, and urinary continence, described as Youssef's syndrome. This patient presented with this classical clinical features. Previously, she had two transverse lower segment cesarean sections. Youssef  in his maiden publication on the syndrome mentioned lower segment cesarean section as the main iatrogenic cause of the condition. Other related risk factors include insufficient dissection of the bladder from the lower uterine segment, excessive intra operative bleeding, use of vacuum for operative vaginal delivery, manual removal of placenta, endometriosis, inflammatory bowel disease, bladder tuberculosis, and congenital lesions.,
In a situation whereby the diagnosis of VUF is early and the fistula is presumed to be small, conservative measures with prolonged catheterization for about 4 weeks of both the cervix and the urethra have been reported to be effective and provides a cure rate in about 5% of cases., Our patient had prolonged urethral catheterization but unfortunately because of the large size of the fistula, spontaneous closure was not achieved. The gold standard for diagnosis is hysterosalpingography (HSG)., In this case, history of two lower segment cesarean sections, classical features of type I Youssef's syndrome  and the HSG findings of continuous opacification of the urinary bladder through the uterine cavity were enough to make the diagnosis. Other diagnostic tools for VUF include the use of cystoscopy and cystography, which would show the bladder and the uterus filled with radio opaque dye indicating communicating pathway between the two organs, and the noninvasive techniques, such as magnetic resonance imaging (MRI). Patients with VUF at times present with urinary tract infection. In this case, she presented with urinary tract infection, which was treated in the hospital the patient was referred to us.
The definitive treatment for Youssef's syndrome is surgical; however, conservative measures such as prolonged catheterization , and medical management to cause induced amenorrhea to aid in fistula healing, like the use of oral contraceptives, progestational agents, and gonadotropin-releasing hormone analog for the induction of amenorrhea, have been reported in the literature with successful outcomes.,
Surgery can be performed transabdominally, endoscopically, and robotically. Transvaginal approach is less favored because of higher location of the fistula and its complexity. In addition, the edge of the fistula could involve the ureteric orifice, which subject the surgery at the risk of ureteral entrapment in the sutures.
The clinical findings were discussed with her, including the possible diagnosis and the planned procedure. She consented for the surgery.
The supra trigonal vesicovaginal fistula repair by modified O'Connor's technique  was employed in this repair. There was no menouria after repair, and menses was commenced normally.
| Conclusion|| |
Youssef's syndrome is a rarely encountered urogynecological fistula. However, with increasing rate of repeat cesarean deliveries, doctors need to be aware of this complication as an important risk is associated with repeat cesarean deliveries. They would also need to discuss this risk when consenting patients for repeat cesarean deliveries. Furthermore, it would be reasonable to ensure that the most senior doctor who has been trained to proficiency performs repeat cesarean deliveries as a precaution to prevent VUF and other morbidities associated with this procedure.
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
Conflicts of interest
There are no conflicts of interest.
| References|| |
Birge O, Ozbey EG, Erkan MM, Arslan D, Kayar I. Youssef's syndrome following caesarean section. Case reports. Obstet Gynecol 2015;2015:1-4.
Eisenkop SM, Richman R, Platt LD, Paul RH. Urinary tract injury during caesarean section. Obst Gynecol 1982;60:591-6.
Buchholz NP, Daly-Grandeau E, Huber-Buccholz MM. Urological complications associated with caesarean section. Eur J Obstet Gynecol Reprod Biol 1994;56:161-3.
Yossepowitch O, Baniel J, Livne PM. Urological injuries during cesarean section: Intraoperative diagnosis and management. J Urol 2004;172:196-9.
Iloabachie GC, Njoku O. Vesico-uterine fistula. British J Urol 1985;57:438-9.
Tancer ML. Vesico uterine fistula‒a review. Obstet Gynaecol Surv 1986;41:743-53.
Eogan M, McKenna P. Conservative management of a traumatic uterovesical fistula ('Youssef's syndrome'). Europ J Obstet Gynecol Rep Biol 2003;110:114-6.
Bhattacharjee S, Kohli UA, Sood A, Tripathy S, Gupta, M. Vesicouterine fistula: Youssef's syndrome. Med J Armed Forces India 2015;71:175-7.
Knipe WHW. Vesico-uterine fistula. Am J Obstet Gynecol. 1908;57:211-7.
Youssef AF. Menouria following lower segment caesarean section: a syndrome. Am J Obstet Gynecol. 1957;73:759-67.
Józwik M., Józwik M. Clinical classification of vesicouterine fistula. Int J Gynecol Obst. 2000;70:353-7.
Shanmugasundaram R, Gopalakrishnan G, Kekre NS. Youssef's syndrome: Is there a better way to diagnose? Indian J Urol 2008;24:269-70.
] [Full text]
DiMarco CS, DiMarco DS, Klingele CJ, Gebhart JB. Vesicouterine fistula: A review of eight cases. Int Urogynecol J Pelvic Floor Dysfunct 2006;17:395-9.
Alkatib M, Franco AV, Fynes MM. Vesicouterine fistula following cesarean delivery: Ultrasound diagnosis and surgical management. Ultrasound Obstet Gynecol 2005;26:183-5.
Kilinc F, Bagis T, Guvel S, Egilmez T, Ozkardes H. Unusual case of post-cesarean vesicouterine fistula (Youssef's syndrome) Int J Urol 2003;10:236-8.
Bastakoti R, Saha R. Conservative management of uterovesical fistula following primary caesarean section. JKMC 2014;2:211-3.
Netto NR, Santos PQ. Youssef's syndrome.
Int Urol Neph 1975;7:115-8.
Rubino SM. Medical treatment of utero-vesical fistula. Lancet 1977;309:900-0.
Yip SK, Leung TY. Vesicouterine fistula: an updated review. Int Urogynecol J Pelvic Floor Dysfunct 1998;9:252-6.
Dalela D, Ranjan P, Sankhwar PL, Sankhwar SN, Naja V, Goel A. Supratrigonal VVF repair by modified O'Connor's technique: An experience of 26 cases. Eur Urol 2006;49:551-6.
[Figure 1], [Figure 2], [Figure 3], [Figure 4]