The incidence of VUF is 1%-4% and its incidence is being increased due to more frequent use of cesarean section. In modern obstetrics VUF usually follows after lower segment cesarean section and accounts for 83% of the cases and rarely follows other causes like vaginal birth after cesarean section, high forceps delivery, and prolonged labor. Youssef in 1957 described the least common type of VUF which presents with classical triad of amenorrhea, cyclic hematuria (menouria) and absence of urinary incontinence which occurs due to inadvertent injury to the bladder during cesarean sections. A majority of these patients are continent of urine and a high index of suspicion is required for its diagnosis. Although it is an uncommon syndrome, current evidence suggests an increasing prevalence of this syndrome due to the higher rate of cesarean section.
A 28 years old, Para 4, Living 2, fresh stillbirth 2 with previous two cesarean sections presented to the out-patient department with complaints of cyclical hematuria with amenorrhea for 10 months which started 6 months after her surgery for rupture uterus which was done for obstructed labor in a remote village. The patient gave history of lactational amenorrhoea for a period of 6 months following which she developed menouria. She continued to take treatment at a private clinic in her village for her complaint. She gave history of prolonged labor and shock for which she was shifted to the operation theater, details of the surgery were not available but she was very sure of uterine rupture. Her postoperative course was uneventful and she was discharged after 10 days. There was no history suggestive of urinary incontinence. She had two vaginal deliveries at home which were fresh still births and two cesarean section following that. On clinical examination she was averagely built and nourished. There was no abnormality detected on general and physical examination. On abdominal examination a vertical midline scar of previous cesarean section was present. On speculum examination there was no fistula seen and there was no urinary incontinence. On per vaginal examination the uterus was anteverted and normal in size. Three swab test was done after injecting methylene blue in the bladder and none of the swabs were stained blue. On checking her previous case records she had reports of pelvic ultrasonography which was suggestive of a cervical mass infiltrating the urinary bladder anteriorly. A computed tomography (CT) was also done, which showed heterogeneously enhancing mass in cervical region with central necrosis infiltrating the body of uterus and upper vaginal wall suspicious of cervical carcinoma with indentation of posterior wall of urinary bladder with mild parametrial fat stranding. In view of these reports she was referred to oncology center for further evaluation. At oncology center Human Papilloma Virus – DNA test was done which was negative. A cystoscopically directed cervical biopsy of the lesion which was situated in the posterior wall of the bladder closed to the bladder
trigone was done. It was suggestive of benign endocervical polyp with squamous metaplasia and no mass lesions in bladder, vagina and cervix. Repeat cystoscopy with vaginoscopy with bladder dye study was done which showed 3× 3 cms vesicouterine fistula in posterior wall close to bladder trigone. On intravesical bladder dye instillation, dye was seen coming through the cervix and vagina. Magnetic resonance imaging (MRI) showed 1.6 cm defect in lower uterine body and posterior bladder more near the left trigone wall suggestive of VUF and uterus, endometrium, vagina and both ureters were otherwise normal. Cervical malignancy was thus ruled out by oncologist and hence the patient was referred to our tertiary care center. Her preoperative laboratory investigations were normal. Urine routine microscopy was normal. Urine culture showed no growth. Hysterosalpinography (HSG) showed streak of contrast passing into the urinary bladder through fistulous track between lower uterine segment and bladder suggesting VUF. MRI plates were reviewed which confirmed the diagnosis of VUF. History and imaging confirmed the diagnosis of Youseff’s syndrome.
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