Uterocutaneous Fistula – Uncommon Presentation of Common Surgeries

The fistula is defined as an abnormal tract communicating two epithelial surfaces, which may be between two lumen or lumen to the exterior surface. Uterocutaneous fistula is a rare pathology where there is abnormal communication between the uterine cavity and skin. The exact incidence is not very well known. Only 120 cases have been described in the literature over the past 200 years.[] The condition is said to be less common compared to uterovesical, uterorectal, uteroperitoneal, and uterovaginal fistulas.[] Common causes of uterocutaneous fistula are surgeries more common with a cesarean, especially with a classical cesarean, myomectomies, hysterotomies, endometriosis, chronic infections such as tuberculosis and actinomycosis, septic abortions, foreign bodies, intrauterine devices, prolonged use of drains, incomplete placental removal, and prolonged labor. Malignancies can cause uterocutaneous fistula due to local invasion or postradiation.[,] Because of the few cases reported in the literature, there are no diagnostic and management guidelines. We report three cases of uterocutaneous fistula, two following cesarean and one following myomectomy, which was managed successfully by surgical excision. CASE REPORTS Case 1 A 29-year-old P3 L1 A1 female underwent elective cesarean at 39 weeks of gestation for postcesarean pregnancy with breech presentation. She had a history of previous 2 term intrauterine fetal death and gestational trophoblastic neoplasia in a previous pregnancy and had completed chemotherapy. During cesarean, dense adhesions were present between the uterus, anterior abdominal wall, and bladder. Adhesiolysis was done. The uterus was closed in a single layer with No. 1 Polyglactin. The rectus sheath was closed with a polydioxanone (PDS) loop and the skin with Monocryl. She was discharged on postoperative day 4. She reported on postoperative day 11 with discharge from skin incision. Blood-tinged serous discharge was present coming from a single defect 1 cm × 0.5 cm above the incision site, which increased in quantity on lying prone [Figure 1]. The culture showed no growth. On clinical suspicion of fistula, magnetic resonance imaging (MRI) was done, which showed a fistulous tract between the lower segment of the uterus to the skin overlying the scar site [Figure 2]. There was a collection 4.6 cm × 4.5 cm within the uterus and 2.1 cm × 6.0 cm collection in the vesicouterine pouch. Four weeks after the cesarean, she was taken for surgical exploration. Hysteroscopy showed submucous fibroid and blood clots. Laparoscopy showed the uterus adhered to the anterior abdominal wall. Bowel and omental adhesions were seen over the uterine surface. Laparotomy was done by Pfannenstiel incision. Adhesiolysis was done. The fistulous tract was excised completely from the skin to the uterus [Figures 3 and 4]. The uterine defect was closed with Polyglactin No. 1. The vascularized omental patch was fixed over the uterine defect. The rectus sheath was closed with PDS, and the skin was closed with silk. Histo Pathological Examination (HPE) showed a fistulous tract with granulation tissue comprising numerous neutrophils, lymphocytes, plasma cells, histiocytes, and foreign body giant cells. No dysplasia, malignancy, or granulomas was seen. Postoperative recovery was good, and at present, she is asymptomatic. Case 2 A 35-year-old A2 female underwent myomectomy 1 year before. An 8 cm × 7 cm intramural fibroid was removed from the fundus which showed cystic degeneration and adenomyotic changes. She had a surgical site infection in the postoperative period and underwent secondary suturing. After 6 months of surgery, she presented with discharge from the laparotomy scar site during menses. MRI done showed a fistula connecting the fundus of the uterus to the subcutaneous tissue [Figures 5 and 6]. She underwent exploratory laparotomy. The abdomen was opened by a previous Pfannenstiel incision. The fistulous tract was seen from the skin traversing through the rectus sheath to the fundus of the uterus. The uterus adhered to the anterior abdominal wall. Adhesiolysis was done. Complete excision of the fistulous tract from the skin to the uterine fundus was done [Figures 7-9]. The uterine fundus was repaired with No. 1 Polyglactin. Vascularized omental patch attached to the fundus over the uterine scar. The rectus sheath was closed with PDS, and the skin was closed with Monocryl. Histopathology showed a fistulous tract with endometriosis. Postoperative recovery was good. At present, patient is asymptomatic and comfortable. Case 3 A 32-year-old P3 L2 A3 female underwent elective lower-segment cesarean section (LSCS) for the previous 2 cesarean at 39 weeks of gestation. Intraoperatively, she had dense adhesions between the uterus and the anterior abdominal wall. Adhesiolysis was done. The uterus was closed with Vicryl No. 1 in a single layer. She presented to us 6 weeks after LSCS with blood-tinged serosanguinous discharge. MRI showed a fistula connecting the lower segment of the uterus to the scar site. She underwent laparotomy and excision of the fistula. The uterus was repaired with polyglactin. HPE showed a fistulous tract with fibrosis. No dysplasia, malignancy, or granulomas was seen. Her postoperative recovery was good, and at present, she is asymptomatic.

Although uterocutaneous fistula is not commonly seen in our practice, it can cause significant morbidity to patients. Clinical diagnosis is possible when there is persistent chronic discharge postsurgery, which may or not be associated or exaggerated during menses. The pathophysiology is not well understood. Predisposing factors are repeated cesareans and surgeries, incomplete and inappropriate closure of incisions, less blood supply hampering wound healing, anemia, poor nutritional status, hypoproteinemia, suture line necrosis due to deficient blood supply which may happen due to excess use of electrocautery, very tight hemostatic sutures, and inappropriate use of nonabsorbable sutures. There is no consensus regarding the best investigation for diagnosis. Ultrasonography may show the tract sometimes. Fistulogram and contrast-enhanced computed tomogram help in diagnosis. A contrast dye may be injected through the cervix, and hysterosalpingography performed may show the fistula. Hysteroscopy may or may not show the fistula. MRI due to better soft-tissue enhancement may delineate the fistulous tract better and help us plan surgery better.[] MRI may also show any associated abscess, anatomical variation, and activity of the disease. Due to noninvasiveness, absence of radiation, and better soft-tissue delineation, MRI is a good diagnostic modality if cost is not an inhibiting factor. Intraoperatively, the fistulous tract may be delineated using methylene blue instillation through the skin, or by probing through a small Hegar dilator. Treatment may be conservative, medical, or surgical. Successful healing of the fistula has been reported with gonadotropin-releasing hormone (GnRH) analogs by a few authors. GnRH agonists by stopping menses and causing atrophy of endometrium decrease the discharge leading to healing of the fistula.[] Complete surgical excision is the preferred surgical management for uterocutaneous fistula, which can be done by laparotomy as well as by laparoscopy in expert hands.[,] Earlier, hysterectomy has been performed for uterocutaneous fistulas; however, such radical treatment may not be necessary in the present day with good surgical techniques and good suture material. Excision of the tract with adequate margins, complete debridement of necrotic tissues, and maintaining good vascularity of the margins will help better healing, and chances of recurrence will be minimized. The role of adhesion barriers like placing a vascularized omental patch or placing oxidized regenerated cellulose is not yet well studied in preventing recurrence. Antibiotic cover, drainage of abscess, good wound care, nutritional optimization of the patient, and correcting anemia and hypoproteinemia will help in wound healing. Combined medical and surgical treatment reducing the discharge by GnRH agonists followed by surgical excision may be tried. It may also buy time to build up the patient to adequate perioperative conditions medically. Endoscopic surgical excision is possible with adequate expertise. Further studies on the pathogenesis of fistula may make medical treatment possible if it is hormone dependent or tumor necrosis factor-alpha dependent as in inflammatory bowel disease.[] Local injection of mesenchymal stem cell therapy can be an exciting option as in the perianal fistula of Crohn’s disease. Uterocutaneous fistula can be prevented by preventing infections, good surgical practices, optimizing perioperative nutritional status of the patient, and avoiding multiple surgeries.

Uterocutaneous fistula is rare. It is best to prevent it by reducing postoperative infection and by following the best surgical practices. MRI is a safe and reliable modality to diagnose uterocutaneous fistula. Surgical excision by laparotomy or laparoscopy is safe and provides a complete cure and preserves fertility. 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.

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