Successful medical management of uterocutaneous fistula

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A 28-year-old woman (P2L2) presented to the Department of Obstetrics and Gynecology at Dr Ram Manohar Lohia Hospital, New Delhi, India, in May 2012 with persistent purulent discharge from an opening in the Pfannenstiel scar after her second cesarean delivery (carried out in December 2011 at a private hospital). According to the patient's discharge summary, it was a difficult surgery; her uterus and rectus abdominis were densely adherent and sharp dissection was required to create a space for delivery of the infant. Excessive intraoperative bleeding ensued, requiring 2 units of transfused blood. An intraperitoneal drain was placed and the abdomen closed. The woman was discharged in satisfactory condition but reported back after 1.5 months with persistent purulent discharge from the stitch line. Abdominal ultrasound was performed to rule out pelvic collection; it revealed the uterus to be adherent to the anterior abdominal wall and revealed a well-defined linear tract extending from the anterior uterine wall to the skin. Magnetic resonance imaging corroborated the ultrasound findings (Fig. 1). A fistulogram further confirmed the diagnosis (Fig. 2). Magnetic resonance image showing uterocutaneous fistula (arrow). Fistulogram showing fistula (arrow). On examination in May 2012, her abdomen was soft and there was an opening (5 × 5 mm) in the center of the Pfannenstiel scar exuding pus. Speculum examination revealed the cervix to be healthy but "pulled up." Vaginal examination revealed the uterus to be of normal size but it was noted to be pulled up above the pubic symphysis, indicating adherence to the anterior abdominal wall. Uterine mobility was restricted and painful. Bilateral fornices were free and non-tender. The pus was sent for culture and sensitivity, and broad-spectrum antibiotics were started. Examination during menstruation showed menstrual blood oozing through the fistulous opening, which is pathognomonic of uterocutaneous fistula. The woman was counseled for hysterectomy and excision of the fistulous tract. She was keen to preserve her uterus, so a decision was taken—with the consent of the patient—to administer gonadotropin-releasing hormone (GnRH) analog in the hope of spontaneous closure of the fistula. In the case of treatment failure, surgery would be performed. Intramuscular triptorelin (3.75 mg) was injected at 4-weekly intervals for a total of 6 doses. During this period, the patient remained amenorrheic and the size of the fistulous opening decreased until it closed completely after the fifth dose. The sixth dose was administered to sustain amenorrhea for longer; the woman resumed menstruation 3 months after the last injection and has had regular menstrual cycles since then. No further discharge has been noted and the fistula has remained closed. Uterocutaneous fistula is a very rare complication, with a literature review finding only 18 case reports; medical management was attempted in only 2 of these cases [1,2], with successful closure reported in only 1 case [1]. In the remaining cases, surgical management in the form of hysterectomy and excision of the fistulous tract was performed [3,4]. Review of the literature also revealed possible etiologies: history of multiple abdominal surgeries [1,4]; use of drains; incomplete closure of uterine wound during cesarean delivery; and intra-abdominal sepsis in the presence of previous abdominal scar. Induced abortion with uterine perforation, migration of an intrauterine device, and secondary abdominal pregnancy have also resulted in uterocutaneous fistula. In the present case, the probable cause of uterocutaneous fistula was repeat cesarean in the presence of dense adhesions between the uterus and the abdominal parieties. It is possible that, in the rush to deliver the infant and owing to dense adhesions and excessive bleeding, the proper plane could not be delineated, leading to improper closure of the uterine wound. Hematoma and collection from the deficient uterine wound may have promoted sepsis, leading to discharge of the purulent collection through the weak incisional area and formation of uterocutaneous fistula. Because this complication is rare, a set protocol for optimal management is not clear. Ideally, the condition would be avoided; it should be anticipated in all cases in which a previous cesarean delivery has been performed and in which there is suspected uterine adherence to the anterior abdominal wall. For all such patients, the next surgery should be performed by an experienced surgeon. Precautions must be taken in the form of the abdomen being opened with a longitudinal incision, starting from the adhesion-free area, and delineating the proper plane. If unable to lyse the adhesions, upper segment cesarean is a reasonable option. The mechanisms of action of GnRH agonists in the management of fistula are not clear. However, cessation of menstruation, endometrial atrophy, and decreasing uterine size may cause cicatrization and contracture of the fistulous tract, leading to fibrosis and closure. Because prolonged treatment with GnRH has its own limitations, patients should be reviewed after 6 months for failure of medical management. Medical therapy with GnRH agonists is a cost-effective and uterus-conserving management option and should be attempted before resorting to surgical management for uterocutaneous fistula. The authors have no conflicts of interest.