Utero-cutaneous fistula following caesarean section.

A multiparous woman in her 20s, presented to the clinic with bleeding from caesarean scar site. After clinical examination and radiological evaluation with ultrasound and MR fistulogram, the diagnosis was utero-cutaneous fistula. Complete resection of fistulous tract with uterine defect closure and supportive omental patch placement was done after an intraoperative demonstration of the utero-cutaneous fistula by injecting methylene blue dye. Utero-cutaneous fistula is a rare complication after caesarean section. Cyclical bleeding from an abnormal opening in previous caesarean scar site is the classical presentation of this condition.

Obstetrics and gynaecology, Surgery

Utero-cutaneous fistulae are so rare that only 120 cases are reported in the literature.1 Diagnosis can be achieved with MR fistulogram and prompt surgical excision of tract, repair of defect and omentoplasty will give excellent results postprocedure. Case presentation A multiparous woman in her 20s, para 2 with two previous caesarean deliveries, presented to the clinic with bleeding from a point just above the caesarean scar site in the abdomen. The last caesarean section was done 5 months ago. She resumed her regular menstrual cycles 4 months after delivery. Her last menstrual period was 5 days prior to the presentation. On examination, she was thin-built with a body mass index of 21. Her general examination was within normal limits. Abdomen examination showed two tiny reddish spots (figure 1). One spot was just above the scar near the midline. The other spot was an exuberant granulation tissue. It was non-tender on palpation and no mass was felt. The patient was asked to come back during menstrual cycles and a provisional diagnosis of scar endometriosis was made. She was reviewed in the next cycle with bleeding from the first point and the other granulation tissue remained the same with no bleeding. An ultrasound was done which raised a suspicion of a fistulous tract extending from anterior surface of uterus to the superficial skin. This was confirmed with an MR fistulogram (figure 2). The fistulogram revealed a 9 mm defect in the anterior wall of the uterus, just above the cervix communicating with the exterior at the caesarean scar site through a fistulous tract. The fistulous tract measured 27 mm. Treatment A general surgeon’s assistance was sought to reconstruct the abdominal wall in the event of a large defect postexcision. The tract was identified during surgery by instilling methylene blue into the tract through the cervix (figure 3). After identification with dye, a probe was passed through the tract abdominally and the abdomen was opened in layers around the tract (figure 4). The tract was then completely excised and the defect in the uterus was closed in two layers. It was further supported with an omental patch. Bilateral tubal ligation was done as requested by the patient. As the abdominal wall defect was not too large, an anatomical closure was done. Outcome and follow-up Bleeding during menstruation at the previous surgical scar stopped postsurgery. Regular menstruation continued without the discharge at anterior abdominal wall.

A fistulous tract is an abnormal communication between two epithelium-lined surfaces. It usually results from an injury due to trauma or infections. Obstetric fistulae usually are urinary fistulae and rarely can be utero-colonic. However, now with increasing rates of caesarean section, utero-cutaneous fistulas are increasing. Though rare, it is being reported. Risk factors include multiple abdominal surgeries that involve entry into the uterine cavity not just caesarean section, but procedures like myomectomy, hysterotomy, etc., seem to be associated with utero cutaneous fistula.2–5 Especially so, if the postoperative period is complicated with infection. Probably infection and over-suturing leads to sloughing of the anterior uterine wall, leading to formation of a fistula. Undiagnosed or partially treated Mullerian anomaly has also led to development of utero-cutaneous fistula.6 There was one case report of a woman developing fistula, secondary to closure of the uterus with silk, a non-absorbable suture material, which would have been a nidus for infection.5 The symptoms are usually cyclical swelling and bloody discharge, which can mimic an endometriotic nodule of the scar. Imaging helps to differentiate between the two. Various imaging techniques are used. However, a fistulogram with either CT or MRI in the sagittal sections would give a clear diagnosis.7 Hysteroscopy helps us visualise the inner opening of the fistula. In lower resource settings, injection of methylene blue into the cervix, would confirm the diagnosis as done in the present case and also reported in the literature.8 Attempting to excise the endometriotic nodule without realising it as a fistulous tract has led to hysterectomy in a case report.9 So, imaging is important before starting treatment. Reviewing the literature shows that surgery remains the mainstay in the management of utero-cutaneous fistula.10 All case reports suggest excision of the tract. After excision of the tract, the uterus needs to be closed in two layers with absorbable sutures. However, before planning excision, appropriate control of infection needs to be achieved. Removal of the uterus may or may not be done, depending on the degree of involvement. Medical management with gonadotropin agonists has been shown to be effective in small fistulae.11 12 In conclusion, women who present with bleeding from the scar site must be thoroughly evaluated before starting a treatment protocol. Also, early surgical intervention should be the mainstay of treatment to avoid patient distress and hysterectomy. Learning points. Recurrent presentations of discharge during menstruation at the site of caesarean scar or nearby, must raise high suspicion of utero-cutaneous fistulae. Early diagnosis with MR fistulogram and surgical intervention could cure the ailment with good results. Better surgical technique of caesarean section could lower the incidence of utero-cutaneous fistula in immunocompetent patients. Footnotes Contributors: SC, IG, VND, SG were responsible for drafting of the text, sourcing and editing of clinical images, investigation results, drawing original diagrams and algorithms, and critical revision for important intellectual content. SC, IG, VND, SG gave final approval of the manuscript. Funding: The authors have not declared a specific grant for this research from any funding agency in the public, commercial or not-for-profit sectors. Case reports provide a valuable learning resource for the scientific community and can indicate areas of interest for future research. They should not be used in isolation to guide treatment choices or public health policy. Competing interests: None declared. Provenance and peer review: Not commissioned; externally peer reviewed. Ethics statements Patient consent for publication Consent obtained directly from patient(s).

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