Utero-cutaneous Fistula: Laparoscopic Management of a Rarity - A Case Report

A 27-year-old, para one lady, presented to us 3 months after having undergone a cesarean section at another hospital. The main symptom at presentation was discharge from one corner of the wound for 1 week on a background of low grade fever and painful urination ever since her cesarean section 3 months prior for which she had been prescribed analgesics and antibiotics on and off. At our center, she was initially managed as a case of surgical site infection which involved opening the wound, frequent dressing for a few days followed by re-suturing in the operation theater. Thereafter, the patient was put on antibiotics and anti-inflammatory drugs and discharged. However, on her follow-up visit, it was noticed that the discharge was still profuse and rather, resembled menstrual blood with no resolution of fever or painful urination. The peculiar menstrual blood like resemblance of the discharge prompted us to suspect that it was more sinister than just a surgical site infection. The routine ultrasound (US) suspected an abnormal communication between the uterus and the skin. To confirm the US findings, CECT was done which clearly demonstrated a communicating tract between the uterus and the skin. Considering the high prevalence of tuberculosis in our country, the same was also, ruled out with the basic investigations such as erythrocyte sedimentation rate, tuberculin test, and chest X-ray. Once the work up was complete and after elaborate counseling, the patient was taken up for laparoscopic exploration followed by fistula tract excision. We used one 10-mm supra umbilical port, two 5-mm ports on the left side, and one 5-mm port on the right side. On laparoscopy, we noted a dense band from the anterior uterine wall up to the abdominal wall. The surgical procedure involved meticulous dissection in layers as the tract could contained vessels or visceral parts or be traversing through viscera, as in our case. Gradual dissection of the band revealed the tract within running from the uterus along the bladder dome laterally, right up to the skin [ Figure 1 ]. To facilitate complete tract excision, a deliberate 2 cm cystotomy incision had to be made in the bladder, which was later sutured in layers. The uterine side of the tract was noted to be necrotic, so the uterine edges were freshened and later sutured to close the defect. Hemostasis was maintained at every step. Laparoscopic view of the fistulous tract within the band of adhesion Postoperatively, the patient was put on antibiotics, catheterization for 2 weeks with urinary antiseptics for a month. Histopathology revealed a tract lined by endometrial epithelium with surrounding fibromuscular tissue, reminiscent of “mint with a hole” appearance [ Figure 2 ]. On her 6-month follow-up, she is doing well with no significant complaints. Histopathology displaying the tract lined by endometrial epithelium with surrounding fibromuscular tissue - “Mint with the hole”

A fistula is an abnormal communication between two epithelial surfaces either between two hollow organs or between a hollow organ and surface epithelium.[ 1 ] They are usually lined by granulation tissue that gets epithelized over time leading to distressing symptomatology.[ 2 ] Communications between the uterus and bowel or bladder are more commonly encountered by gynecologists while those between the uterus and skin are rarely encountered. These fistulae occur due to postoperative or postpartum complications such as injuries or infections, use of drains, and incomplete closure of incisions.[ 3 ] Utero-cutaneous fistula (UCF) is a very rare complication of cesarean section with only about 20 reported cases[ 4 ] and being this rare a condition, it requires thorough work up by means of fluoroscopy, contrast-enhanced computed tomography (CECT) or magnetic resonance imaging (MRI) to properly delineate the tract to ensure appropriate management. Although surgical management is the definitive treatment, there are reports of two cases of UCF which were successfully managed using the medical methods.[ 5 6 ] We report this case to highlight that UCF can be successfully managed by laparoscopic route.

UCF are rare, can be difficult to diagnose, cumbersome to manage and should be kept in mind as a differential of nonhealing wounds after cesarean sections and uterine surgeries. Meticulous uterine closure after uterine surgeries and avoiding postoperative infections can prevent future UCF formation. Surgical repair remains the mainstay of treatment despite few reports suggesting the trial of medical management. This study was conducted in accordance with the ethical principles outlined in the Declaration of Helsinki and its amendments. The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient has given her consent for her images and other clinical information to be reported in the journal. The patient understands that name and initials will not be published and due efforts will be made to conceal identity, but anonymity cannot be guaranteed. Amarinder Singh- Helped plan the surgery, was one of the surgical assistants and also, involved in the case report conceptualization; Sampath- Helped in preoperative work up, was one of the surgical assistants and helped in manuscript drafting; Poddar- Helped in critical revision of the manuscript; Rasheed- Was one of the surgical assistants, helped in preoperative work up, manuscript writing and critical revision; Shiv Kumar- Pathologist assisted with final analysis and manuscript writing.; Shikha Sharma- Was main surgeon, study conception, manuscript writing, and follow up. All authors have read and agreed to the final version of the manuscript. Data sharing is not applicable to this article as no datasets were generated or analyzed during the current study. There are no conflicts of interest.

UCF are rare, with only about 20 cases reported in the literature.[ 4 ] The more commonly witnessed fistulae by gynecologists are utero-vesical and utero-colonic. The first case of UCF, as mentioned in the literature, was reported by Todd in 1939 in a young women who had to undergo radical surgery for the UCF occurring after a prior laparotomy for bilateral pyosalpinx.[ 7 ] Owing to its rare presentation, the approach to the diagnosis and management of UCF is blurred. The possible etiologies are multiple abdominal surgeries, incomplete closure of uterine wound during myomectomy or cesarean, use of drains, postoperative infections or inflammation. Furthermore, septic abortions, migration of intra-uterine devices, endometriosis, and retained products of conception have been attributed as rarer possibilities.[ 8 ] Most reported cases were due to a postoperative abscess after a caesarean section or after a curettage for septic abortion leading to uterine perforation.[ 9 ] In our case, it is assumed that postoperative inflammation and infection after the cesarean could have caused epithelial disruption leading to the uterine wall coming in direct communication with the abdominal wall, subsequently, leading to the formation of a fistulous tract. It is usually misdiagnosed with wound infection initially, as in our case. The diagnosis can be confirmed by either a fistulogram, CECT, MRI or methylene blue test.[ 9 ] Considering its rare presentation, there is no standard approach to the management of UCF. Although, literature talks about two cases successfully managed using GnRH agonists, surgical management is accepted as the treatment of choice.[ 5 6 ] Options for surgical management include laparotomy with/without hysterectomy, laparoscopy combined with mini-laparotomy and laparoscopy tract excision. In our case, we opted for laparoscopic exploration followed by fistulous tract excision, debridement of uterine necrotic tissue, and suturing of the uterine defect as well as closure of the deliberate bladder cystotomy defect. We could only find one case successfully managed laparoscopically by Shah et al .[ 10 ] Yesiladali et al . reported a case of postmyomectomy UCF who was managed with combined laparoscopy and mini-laparotomy and later, successfully conceived also.[ 3 ] A successful laparoscopic repair of UCF requires thorough preoperative mapping of the tract using the best available radiological modalities followed by meticulous step wise excision and repair. Our patient on her follow-up is doing well with no significant complaints.