Overview Jejuno-uterine fistula is a very rare occurrence. The case presented described the development of a jejuno-uterine fistula following uterine artery embolization (UAE), which we believe is the first reported case.
Jejuno-Uterine Fistula After Endovascular Embolization For Uterine Bleeding
Simon D. Eiref, MD,1 Scott Holekamp, MD,1 John Koulos, MD,2 Gabriel Levi, MD,3 Marie Winestone, MD,4 Alexander Kagen, MD,4 and I. Michael Leitman, MD1
Departments of Surgery,1 Obstetrics and Gynecology,2 Pathology,3 and Radiology,4 Albert Einstein College of Medicine-Beth Israel Medical Center, New York, New York
Contact: I. Michael Leitman, MD E-mail
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Citation: Eiref SD, Holekamp S, Koulos J, Levi G, Winestone M, Kagen A, and Leitman IM. Jejuno-uterine fistula after endovascular embolization for uterine bleeding. J Surg Radiol. 2010 Oct 1;1(2).
Received June 30, 2010. Accepted July 21, 2010. Epub August 1, 2010. Copyright: © 2010 Surgisphere Corporation. This is an open-access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited. |
Contents - Introduction- Discussion - Disclosures - References |
Introduction
Jejuno-uterine fistulas have rarely been described complications of curettage procedures. We describe the first case report of a jejuno-uterine fistula, which formed as a result of uterine artery embolization.
Case Report
A 37 year old Asian female, G0P0, with a history of adenomyosis and endometrial hyperplasia, presented to the hospital with prolonged heavy vaginal bleeding and anemia. She underwent hysteroscopy, dilatation and curettage. Following the procedure, brisk hemorrhage from the uterine cavity lead to transfer to the interventional radiology suite. Definitive control of the bleeding was achieved by bilateral uterine artery embolization using Embospheres (BioSphere Medical, Inc. Rockland, MA). Although the recommendation was made for the patient to undergo a scheduled hysterectomy after discharge, the patient was lost to follow-up.
Three months later, she returned to the hospital with complaints of a new onset of lower abdominal pain and painful vaginal discharge. The vaginal discharge increased markedly after eating and contained partially digested food particles. Physical exam was remarkable for a tender lower abdominal mass. CT scan of the abdomen/pelvis (Figure 1) confirmed a jejuno-uterine fistula.
Figure 1. Coronal-oblique multiplanar reconstruction demonstrates fistulous connection between a loop of small bowel and the uterus (arrow). There is layering debris and air in the uterus (arrowhead).
The patient underwent exploratory laparotomy, in which small bowel loops were found to be densely adherent to the fundus of the uterus. Dissection of the small bowel from the uterus revealed a jejuno-uterine fistula between a loop of jejunum (50 cm distal to the ligament of Treitz) and the uterine fundus (Figure 2). The uterine fundus appeared necrotic and ruptured in the midline, appearing as a wedge-shaped defect. The jejunum involved in the fistula was resected and intestinal continuity was reestablished with a primary small bowel anastomosis. Downstream, an 11 x 10 x 3.5 cm partially obstructing foreign body was palpable in the lumen of distal ileum, and was extracted via a separate enterotomy (Figure 3). A total hysterectomy was performed (Figure 4) with an unremarkable postoperative course.
Figure 2. Intraoperative photograph of jejuno-uterine fistula.
Figure 3. Intraoperative photograph of foreign body in small bowel distal to fistula.
Figure 4. Intraoperative photograph of the uterus. The uterine fundus appeared necrotic and ruptured in the midline, appearing as a wedge-shaped defect.
Pathology revealed a 2.5 cm diameter fistulous tract arising from an area of infarcted uterine myometrium to a segment of otherwise normal appearing jejunum. Embolization material was detected within the arteries of the infarcted myometrium, and more interestingly, found extending into the fistulous tract (Figure 5). Analysis of the foreign body removed from the lumen of the distal small bowel revealed that it was infarcted uterine myometrium, matching the wedge-shaped defect in the uterine fundus. There was no evidence of malignancy.
Figure 5. Embolization material is embedded within the inflammation of the small bowel fistulous tract.
Discussion
A fistula is an abnormal connection between two epithelium-lined organs or vessels that do not connect. In the case of the uterus, fistulas can develop to the bladder, colon, and small intestine. Enterouterine fistulas occur infrequently. Martin et al.1 published perhaps the largest review of enterouterine fistulas in 1956 which described 80 cases, 42 of which, followed obstetric injury, 17 resulting from inflammatory processes, 12 following curettage, and 9 related to carcinoma.
In particular, jejuno-uterine fistulas are rare. We found only 4 case reports of jejuno-uterine fistula in the modern literature, each following curettage procedures, and involving the uterine fundus. McFarlane et al.2 described a jejuno-uterine fistula that developed two weeks after dilation and curettage performed for severe postpartum hemorrhage. Duttaroy et al.3 described symptoms of a jejuno-uterine fistula developing 3 months after dilation and evacuation for a spontaneous abortion. Singh et al.4 described a chronic jejuno-uterine fistula following termination of pregnancy, discovered after 3 consecutive abortions. Lastly, Vohra et al.5 described a jejuno-uterine fistula occurring 6 weeks after curettage for retained products of conception. All cases were managed by surgical repair.
The role of uterine artery embolization (UAE) in causing jejuno-uterine fistula has not been previously reported. Nonetheless, significant complications have been described resulting from UAE including uterine necrosis, uterine rupture, and fistulization. Godrey and Zbella6 reported on diffuse uterine necrosis with extensive small bowel adhesions occurring 2 months after UAE of a large uterine leiomyoma. Shashoua et al.7 discussed a case of ischemic uterine rupture occurring 3 months after UAE for symptomatic uterine myomas. Ogliari et al.8 reported on several patients developing uterine cavity defects with cavity-myoma fistulas after UAE for symptomatic leiomyomas. In addition, Sultana et al.9 reported on a vesicouterine fistula developing three months after UAE for myoma.
In our case, the jejuno-uterine fistula followed both curettage and UAE. While curettage appeared to be the etiology of previously reported jejuno-uterine fistulas, we believe that the UAE was the etiology in this case. This was based on the embolization material found by pathology in the fistula tract and the necrotic myometrium discovered distally in the jejunum during surgery.
We suspect that UAE led to necrosis of the uterine fundus in this patient, who had a large uterus and likely poor collateral blood supply. In turn, the necrotic myometrium caused adhesions to the surrounding jejunum, inflammation, and subsequent fistula formation. The presence of foreign body embolization material in the fistula tract would have made spontaneous closure of the fistula unlikely. This mechanism of fistula formation after UAE is in distinction to fistulas developing after curettage, in which the mechanism likely results from direct mechanical perforation of the uterus and possible concurrent injury to adjacent bowel.
Management of jejuno-uterine fistula has traditionally been surgical involving resection of the fistula tract and involved structures followed by primary anastamosis. Recently Józwik10 suggested that endometrium in the fistula tract may be amenable to hormonal regulation and therefore conservative treatment. The embolization material acting as a foreign body in our fistula tract made a conservative approach less attractive. The embospheres were too small to be seen during the resection but were confirmed by pathology. Our surgical approach was similar to ones described by McFarlane, Duttaroy, and Martin. Due to the large defect, we did not attempt to repair the uterus.
Disclosures
The authors have no disclosures or conflicts of interest related to this manuscript.
References
- Martin DH, Hixson CH, Wilson EC Jr. Enterouterine fistula; review; report of an unusual case. Obstet Gynecol. 1956 Apr;7(4):466-9. PubMed
- McFarlane MEC, Plummer JM, Remy T, Christie L, Laws D, Richard H, Cherrie T, Edwards R. Coward C. Jejunouterine fistula: a case report. Gynecol Surg (2008) 5:173–175. CrossRef
- Duttaroy D. Jejuno-uterine fistula. European Journal of Obstetrics & Gynecology and Reproductive Biology 129 (2006) 92–99. CrossRef
- Singh RB, Pavithran NM, Parameswaran RM, Sangwan K. Chronic jejuno-uterine fistula: an unusual cause for recurrent second trimester abortions. Aust N Z J Obstet Gynaecol 2005 45(6);533-534. CrossRef PubMed
- Vohra PA, Kumar Y, Raniga S, Vaidya V, Verma S, Mehta CA Case Of Jejunouterine Fistula Ind J Radiol Imag 2005 15:4:427-428.
- Godfrey CD, Zbella EA. Uterine Necrosis After Uterine Artery Embolization for Leiomyoma. Obstet Gynecol 2001;98:950–2. CrossRef
- Shashoua AR, Stringer NH, Pearlman JB, Behmaram B, Stringer EA. Ischemic uterine rupture and hysterectomy 3 months after uterine artery embolization. J Am Assoc Gynecol Laparosc 2002;9:217–220. CrossRef
- Ogliari KS, Mohallem SV, Barrozo P, Viscomi F. A uterine cavity–myoma communication after uterine artery embolization: two case reports. Fertility and Sterility Vol. 83, No. 1, (2005). CrossRef PubMed
- Sultana CJ, Golberg J, Aizenman L, Chon JK. Vesicouterine fistula after uterine artery embolization: a case report. Am J Obstet Gynecol 2002;187:1726–7. CrossRef PubMed
- Józwik M, Józwik M.Hormonal dependence of fistulas communicating with the uterus. Int Urogynecol J Pelvic Floor Dysfunct. 2007 Jul;18(7):719-20. Epub 2007 Mar 27. CrossRef PubMed
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