Unusual complication after uterine artery embolization and laparoscopic myomectomy in a woman wishing to preserve future fertility

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1 CASE REPORT Unusual complication after uterine artery embolization and laparoscopic myomectomy in a woman wishing to preserve future fertility Olivier Donnez, M.D., Pascale Jadoul, M.D., Jean Squifflet, M.D., and Jacques Donnez, M.D., Ph.D. Department of Gynecology, Universite Catholique de Louvain, Brussels, Belgium Objective: To report a case of uterine fistula arising after laparoscopic myomectomy after a uterine artery embolization. Design: Case report. Setting: A university hospital center. Patient(s): A 38-year-old woman with a uteroperitoneal fistula after laparoscopic myomectomy after a uterine artery embolization. Intervention(s): Laparoscopic excision of the fistula and repair of the myometrial defect with laparoscopic suture. Main Outcome Measure(s): Not applicable. Result(s): Complete correction of the myometrial defect was observed after laparoscopic surgery. Conclusion(s): Uterine artery embolization before myomectomy may interfere with myometrial cicatrization and thus alter the repair. (Fertil Steril Ò 2008;90:2007.e5 e9. Ó2008 by American Society for Reproductive Medicine.) Key Words: Uterine artery embolization, fistula, myomectomy Uterine artery embolization (UAE) for the treatment of uterine fibroids was first performed by Ravina et al. (1) in It is considered to be an option for women with symptomatic fibroids, who are not candidates for surgery or do not wish to undergo an operative procedure (2). The results of UAE are symptomatic improvement of menorrhagia and pelvic pain of around 85% and reduction of myoma size by approximately 50% (3, 4). Complications and secondary effects, such as postembolization syndrome (5), have nevertheless been described. Premyomectomy UAE also has been proposed (6) as a way of reducing operative blood loss and blood transfusion during surgery. However, the procedure has been associated with difficult myoma excision due to loss of the usual surgical plane and the friable texture of myomas (7). Here we describe an unusual complication of UAE followed by laparoscopic myomectomy: a uteroperitoneal fistula that was laparoscopically repaired. Received April 30, 2008; revised May 28, 2008; accepted May 30, O.D. has nothing to disclose. P.J. has nothing to disclose. J.S. has nothing to disclose. J.D. has nothing to disclose. Reprint requests: Jacques Donnez, M.D., Department of Gynecology, Universite Catholique de Louvain, Cliniques Universitaires St. Luc, Avenue Hippocrate 10, 1200 Brussels, Belgium (FAX: þ ; donnez@gyne.ucl.ac.be). CASE REPORT A 38-year-old woman, gravida 0, was referred to our department for a uteroperitoneal fistula after laparoscopic myomectomy performed after UAE. When she was 30 years of age, a uterine myoma was diagnosed in another institution, measuring mm on transvaginal ultrasound. After UAE was performed, myoma size decreased to mm after 2 months. The surgeon then decided to perform a laparoscopic myomectomy 2.5 months after the UAE. The anatomopathological examination showed that a total of 170 g of morcellated myoma has been removed, characterized by ischemia and necrosis. The patient was subsequently asymptomatic. She then presented with 5 years of infertility and underwent laparoscopy and hysteroscopy. Both the fallopian tubes and ovaries looked normal, but when dilute methylene blue was injected through the cervical os, it was visible just beneath the right anterolateral side of the uterine serosa. Hysteroscopy confirmed an intrauterine fistula orifice, originating from the right uterine side wall. The patient was then referred to our department for treatment. Hysterosalpingography revealed a normal-sized uterine cavity and fallopian tubes, but a fistula ran through the right uterine wall (Fig. 1A). Magnetic resonance imagine also was performed, and the transverse view of T2-weighted images confirmed the presence of a right lateral wall fistula through the whole thickness of the myometrium (see Fig. 1B). Because of the possible role of this large fistula in infertility and the increased risk of uterine rupture in case of pregnancy, laparoscopic repair of the uterine defect was proposed to close the fistula. As shown in Figure 2A, laparoscopy easily identified the subserosal defect on the anterolateral side of the /08/$34.00 Fertility and Sterility â Vol. 90, No. 5, November e5 doi: /j.fertnstert Copyright ª2008 American Society for Reproductive Medicine, Published by Elsevier Inc.

2 FIGURE 1 (A) Hysterosalpingography revealed a normal-sized uterine cavity and fallopian tubes, but the presence of a fistula running through the right uterine wall (white arrow). (B) Transverse MRI of T2-weighted images showed the presence of a right lateral wall fistula through the whole thickness of the myometrium (white arrow). uterus. After the serosa had been opened, the external orifice of the fistula was clearly identified. Hysteroscopy confirmed the presence of a 1-cm internal orifice 2 cm beyond the right ostium (see Fig. 2B). Incision of the fibrotic tissue surrounding the fistula was performed by laparoscopy using Metzenbaum scissors (Karl Storz, Tuttlingen, Germany) to identify the external orifice of the fistula (Fig. 3A). Using CO2 laser (LumenisSharplan 1055 S, Yokneam, Israel), we completely excised the fibrotic tissue from the edges of the healthy myometrium to facilitate further healing. A single 2-0 Vicryl SH (Johnson & Johnson, Hamburg, Germany) suture was first placed deep in the myometrium under the mucosa, and then three separate sutures were placed through the entire myometrial thickness. The fistula was finally closed by application of one more 2-0 suture to the serosa. The final view of the repair can be seen in Figure 3B. At the end of surgery, we performed a hysteroscopy to visualize the repair from the uterine cavity. It showed complete closure of the fistula, with the suture just beneath the mucosa. FIGURE 2 (A) Laparoscopy easily identified the subserosal defect on the right anterolateral side of the uterus (white arrow). (B) Hysteroscopy confirmed the presence of a 1-cm internal orifice (black arrows) 2 cm beyond the right ostium (white arrow) e6 Donnez et al. Complications after UAE Vol. 90, No. 5, November 2008

3 FIGURE 3 (A) After opening the serosa, the external orifice of the fistula was clearly identified (black circle). (B) Final view of the repair after closing the myometrium and serosa using separate 2/0 Vicryl sutures. No complications occurred during any of the surgical procedures. The patient was discharged from hospital within 24 hours of surgery. defined, it is particularly useful for women who do not wish to undergo hysterectomy, for those at high surgical risk, including morbid obesity, and for Jehovah s Witnesses (9). Three months after the surgery, we performed another magnetic resonance imaging scan, which showed complete closure and integrity of the right uterine side wall (Fig. 4A). Hysterosalpingography confirmed closure of the fistula (see Fig. 4B). It was recommended that the patient undergo cesarean section in case of pregnancy to avoid the risk of uterine rupture during labor. Nevertheless, UAE is associated with serious complications such as infection, which needs to be distinguished from postembolization syndrome (pain and fever lasting less than 5 days). Infections occur in 1% to 2% of cases (5) and are more frequent with embolization of larger fibroids (10). Cases of necrotic fibroids leading to septic shock and death have been reported (5, 11, 12). To date, five deaths have been documented after UAE among an estimated 50,000 procedures worldwide (13). Approximately 1% to 2% of patients experience ovarian failure after UAE, with most cases occurring in perimenopausal patients (4). Postprocedural hysterectomy is necessary in 1% of patients (2). Uterine artery embolization contributes to adhesion formation (14), and Huang et al. (15) reported pelvic adhesions in DISCUSSION Since Ravina et al. (1) introduced UAE in 1995, this technique has been used to reduce the size of symptomatic uterine myomas and to treat abnormal uterine bleeding. The UAE procedure is cost effective and associated with a short hospital stay (8). Even if the indications for UAE are not yet clearly FIGURE 4 (A) Transverse MRI of T2-weighted images showed complete integrity of the right lateral uterine wall at the site where the fistula was located (white arrow). (B) Hysterosalpingography confirmed closure of the fistula and complete correction of the defect (white arrow). Fertility and Sterility 2007.e7

4 59% of patients who underwent abdominal surgery after failure of UAE. Adhesions are specially associated with embolization of subserosal fibroids, resulting in tissue necrosis and inflammation of the subserosa. Cases of bowel infarction after embolization of subserosal fibroids have also been described (16). In the case presented here, UAE was used to decrease the size of a myoma prior to laparoscopic myomectomy in a patient who wished to preserve her fertility potential. Despite several investigators reporting pregnancies after UAE (17), the use of this technique in women wishing to conceive remains unclear. Indeed, there is a possibility of impairment of myometrial integrity as well as reduction of the ovarian blood supply, leading to a decreased ovarian reserve and premature ovarian failure (18). The true incidence of persistent ovarian failure after UAE is unknown, but has been estimated to be <2% (19). Pregnancies following UAE show higher rates of preterm delivery (odds ratio 6.2; 95% CI, ), malpresentation (odds ratio 4.3; 95% CI, ), abnormal placentation, postpartum hemorrhage, cesarean delivery, and small-for-gestational-age infants compared with pregnancies after laparoscopic myomectomy (17). The investigators concluded that myomectomy should be recommended as the treatment of choice over UAE in most patients wishing to conceive. The Society of Obstetricians and Gynaecologists of Canada recommends that UAE be undertaken as a treatment for fibroids in patients wishing to preserve their fertility, with full disclosure to the patient concerning the limitations of such a procedure and the lack of existing data on future fertility and pregnancy outcome (20). The benefits of UAE before myomectomy are not proven, but the procedure adds significant expense and increases risk (13). Very little information can be found in the literature on the use of UAE before myomectomy. Dumousset et al. (21) reported 22 cases of preoperative UAE and concluded that this procedure does not increase the complication rate or impair operative dissection, and therefore improves the chances of performing conservative surgery. They claim that a multidisciplinary discussion led them to believe that excision of uterine fibroids in three patients would not have been possible without preliminary preoperative UAE, due to the presence of polymyomatous uteri with dominant fibroids larger than 8 cm. This view is strictly subjective, and we believe that polymyomatous uteri are not a contraindication to myomectomy. Ngeh et al. (6) also described preoperative UAE and found a trend toward reduced pyrexia and blood transfusion. This study was small, however, and not a prospective randomized comparison. No information was given in these two studies about the dissection plane of the myomas. Floyd et al. (7) reported a case of abdominal myomectomy after UAE and described a very difficult dissection of the myoma due to degenerative changes. They also recommended that UAE not be used for women of childbearing age, who may desire future pregnancy. Ogliari et al. (22) reported two cases of uterine cavity myoma fistulas after UAE, which were corrected by laparoscopic resection of the myomas. We did not find any large series of UAE before myomectomy. There is a lack of information about its effect on the blood supply in terms of myometrial cicatrization after myomectomy and the integrity of the myometrium. Here, we report an unusual complication arising after UAE and laparoscopic myomectomy. Uterine artery embolization was used in this case to reduce the size of the myoma to facilitate further laparoscopic myomectomy. Myometrial healing after myomectomy was not sufficient, however, to achieve complete integrity of the uterine wall, and a large dehiscence was observed. This suggests that UAE may be associated with a reduced blood supply, and using this technique before myomectomy could interfere with myometrial cicatrization, resulting in a dehiscent scar. This dehiscence could also be a consequence of the technique of laparoscopic myomectomy, but the case we report suggests than an association of these two techniques may be risky for myometrial healing, resulting in this type of lesion. Large series are thus needed to pinpoint the optimal site of UAE before conservative surgery. CONCLUSION Our case suggests that myomectomy performed after UAE may be complicated by a fistula or dehiscent myometrial scar. This complication has never before been described in the literature. Because of possible impairment of the myometrium, we firmly believe that preoperative UAE should be avoided before myomectomy in women who are considering pregnancy. REFERENCES 1. Ravina JH, Herbreteau D, Ciraru-Vigneron N, Houdart E, Aymar A, Merland JJ. 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