Case Report

Uterine Cavity–Myoma Fistula after Hysteroscopic MyomectomyMimicking Uterine Perforation at Hysterosalpingography: CaseReport

Sepideh Peivandi, MD*, Frances R. Batzer, MD, MBE, and Gregory T. Fossum, MDFrom the Divisions of Reproductive Endocrinology and Infertility, Departments of Gynecology and Obstetrics, Mazandaran University of Medical Science,

Faculty of Medicine, Sari, Iran (Dr. Peivandi), and Thomas Jefferson University Hospital, Philadelphia, Pennsylvania (Drs. Batzer and Fossum).

ABSTRACT Fistula formation between the uterine cavity and the cavity of a subserosal myoma was diagnosed at laparoscopy/hysteroscopy

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in a 39-year-old woman with primary infertility. The patient had undergone 2 previous hysteroscopic resection procedures forremoval of a submucosal myoma as part of infertility treatment. Hysterosalpingography demonstrated leakage of contrastmedium from the uterine cavity, a characteristic feature of uterine perforation. At hysteroscopy/laparoscopy, a defect wasobserved in the posterior wall of the uterine cavity with connection to the cavity of a subserosal myoma without any tractto the peritoneal cavity. Laparoscopic myomectomy was performed to repair the uterine wall defect at the site of the fistula.Subsequently, the patient conceived after an office-based gonadotropin cycle therapy and is currently 20 weeks pregnant.Journal of Minimally Invasive Gynecology (2011) 18, 534–537 � 2011 AAGL. All rights reserved.

Keywords: Case report; Hysteroscopic myomectomy; Uterine cavity-myoma fistula

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thor: Sepideh Peivandi, MD, Department of Gynecology

F Center of Emam-Khomeini University Hospital, Sari,

ndi@yahoo.com

y 25, 2011. Accepted for publication April 18, 2011.

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Uterine leiomyomas are the most common pelvic tumorin women, occurring in 20% to 40% of women during theirreproductive years [1,2]. They are frequently associated withsymptoms that affect quality of life, and require treatment[3]. Symptoms usually correlate with the location of themyomas, their size, or concomitant degenerative changes.Leiomyomas are estimated to account for infertility in 1%to 2% of women [1]. Leiomyomas that distort the uterinecavity (submucosal or intramural with an intracavitarycomponent) result in difficulty in conceiving and increasedrisk of miscarriage [4]. Medical treatment of leiomyomasin infertile women attempting to become pregnant is usuallyunsuccessful. Thus, in patients with a submucosal or intra-

mural leiomyoma that deforms the uterine cavity, myomec-tomy is preferred.

Hysteroscopic myomectomy, a minimally invasive surgi-cal procedure, is the treatment of choice for submucosalmyomas and intramural leiomyomas when muchof the myoma protrudes into the uterine cavity [5,6].Complications of hysteroscopic myomectomy are rare. Toour knowledge, there has been no report of fistulaformation between the uterine cavity and the cavity ofa degenerated subserosal leiomyoma after hysteroscopicmyomectomy. Herein we present a case of primaryinfertility in a patient with a history of 2 previoushysteroscopic submucosal myomectomies and subsequentfistula formation between the uterine cavity and the cavityof a subserosal leiomyoma, diagnosed at hysteroscopy/laparoscopy performed after findings mimicking uterineperforation at routine hysterosalpingography.

Case Report

A 39-year-old nulligravida woman was evaluatedbecause of primary infertility of 5 years’ duration. In an

Fig. 1

Hysterosalpingogram shows spillage of contrast medium from fundus

of uterus, mimicking uterine perforation.

Fig. 3

Laparoscopy demonstrated a subserosal myoma in the superior

posterior wall of the uterus and another intramural myoma in the lower

segment.

Peivandi et al. Uterine Cavity-Myoma Fistula after Hysteroscopic Myomectomy 535

attempt to conceive, the patient had undergone 2 previoushysteroscopic resection procedures for removal of a submu-cosal myoma. The first surgery was performed in 2006, andthe second in 2007. Specific documentation from thesesurgeries was not available. Subsequent to the 2 hystero-scopic procedures, hysterosalpingography was performedand demonstrated what was thought to be a fistula with spill-age of contrast medium from the fundal portion seen at thefirst injection of contrast without any pressure (Fig. 1). Thepreoperative diagnosis was perforation of the uterus into theperitoneal cavity due to uterine wall weakness and postsur-gical changes probably at the site of the previousmyomectomies. The patient was taken to the operatingroom for laparoscopy/hysteroscopy and repair of a uterinewall defect.

Fig. 2

Hysteroscopic image shows the opening with well-defined border in the

posterior wall of the uterine cavity near the cornua.

At hysteroscopy, a 2 ! 2-cm defect with well-definedborders was observed on the posterior wall of uterus nearthe left cornua (Fig. 2). The remainder of the cavity wasnormal. At simultaneous laparoscopy, a 4 ! 3 ! 5-cmsubserosal myoma was observed in the superior posteriorwall of the uterus near the fundus, and another intramuralmyoma was located in the lower segment of the uterus(Fig. 3). The remainder of the uterus appeared normal.No connection between the uterine and peritoneal cavitieswas demonstrated using dye injection. The fallopian tubeswere open without any adhesion or endometriosis. After my-omectomy, a fistula from the uterine cavity into a subserosalmyomawas identified. The defect was a fistulous connectionbetween the uterine cavity and the cavity of a subserosal

Fig. 4

Laparoscopic myomectomy and the site of the fistula into the cavity of

the myoma.

536 Journal of Minimally Invasive Gynecology, Vol 18, No 4, July/August 2011

myoma (Fig. 4). Laparoscopic myomectomy and resectionof the edge of the fistula with suturing to correct the uterinewall defect at the site of the fistula were performed. Thepathology report confirmed a myoma. A postoperativehysterosalpingogram revealed a normal uterine cavity. Thepatient underwent intrauterine insemination after gonadotro-pin therapy to induce ovulation. A 20-week singletonpregnancy is ongoing.

Discussion

Hysteroscopic myomectomy is performed to treatintracavitary myomas (i.e., submucosal and intramuralleiomyomas) when much of the myoma protrudes into theuterine cavity. In series of 200 or more hysteroscopicmyomectomy procedures, a complication rate of 0.8% to2.6% has been reported [7,8]. Uterine perforation,distention system hazard, infection, and hemorrhage arethe most common complications. Extensive resectionincreases the risk of uterine perforation; however, thiscomplication is uncommon [9]. There have been no case re-ports of uterine rupture after hysteroscopic myomectomy[6,10]. To our knowledge, this is the first reported case offistula formation between the uterine cavity and the cavityof a subserosal myoma after hysteroscopic resection ofa submucosal myoma.

Apparent myoma necrosis associated with uterinewall defect or fistula formation after uterine arteryembolization (UAE) has been reported [11–16]. Uterineartery embolization is becoming a relatively commontreatment for uterine myomas, with a low risk of severecomplications. The objective of transcatheter embolizationof the uterine arteries is to occlude the vascular supply tothe myoma, causing ischemic shrinkage of the myoma[16–18]. Data about fertility after embolization are scarcebecause most physicians reserve the procedure for use inwomen who do not desire subsequent pregnancy [17].

De Iaco et al [16] reported development of a uterinefistula and discontinuity of the myometrium after hystero-scopic resection of an embolized migrated myoma ina patient who had previously undergone UAE. They specu-lated that this was due to development of an avascularmyometrium after UAE. The patient reported no symptoms;however, routine diagnostic hysteroscopy revealed a 2-cmdiscontinuity of the uterine wall at the site of the previousresectoscopic myomectomy. The myometrium was whiteand thin, and less than normal thickness [16].

Sultana et al [12] reported the first case of a fistula involv-ing the bladder after UAE. A possible explanation for thiscomplication was that the blood supply to the posteriorbladder was affected by the procedure. The superior, middle,and inferior vesical arteries arise from the anterior divisionof the internal iliac artery. Nontargeted embolization orbackflow of the embolization material into the anterior divi-sion proximal to the uterine artery could have occurred [12].

De Iaco et al [11] reported a uterine wall defect thatdeveloped 14 months after UAE in a 54-year-old woman,gravida 1, with a history of pelvic pain and intermenstrualbleeding. A CO2 diagnostic hysteroscopy demonstrateda defect 2 cm wide and 2 cm deep with regular well-defined borders in the right uterine wall. The defect waslimited to the internal myometrium and did not lead to theperitoneal cavity. Rather, it opened into the uterine cavitythrough a 1.5-cm fistula of endometrium and myometrium.The uterine cavity was regular elsewhere, with normalmucosa [11]. Ogliari et al [13] also reported 2 cases ofuterine-myoma communication after UAE diagnosed athysteroscopic follow-up.

Borghi et al [19] reported an ongoing pregnancy afteruterine corrective surgery of a uterine cavity–myoma fistulaafter UAE failure in an infertile woman. An abdominalmultiple myomectomy and correction of the wall defectwas performed. These authors suggested careful study ofthe uterine cavity after UAE failure to detect myometrialabnormalities [19].

Unlike previous cases, our patient had no history of UAE.Thus, the studies cited are not exactly relevant to our casebecause fistula formation into another myoma after hystero-scopic myomectomy has not been previously reported. Itcould be possible that the pathophysiology of UAE fistulaformation and hysteroscopic myomectomy fistula formationare similar in that both result from tissue necrosis. Thermalinjury or muscle wall weakness with resultant elasticity atthe site of our patient’s 2 previous myomectomies couldhave led to a region of discontinuity into the cavity of a sub-serosal myoma.

In conclusion, because hysteroscopic myomectomy iswidely used to treat infertility, it is important to reassessthe uterine cavity at hysteroscopy or hysterosalpingographyin patients with a history of previous hysteroscopic myomec-tomy, to confirm integrity of the cavity.

References

1. Buttram VC Jr, Reiter RC. Uterine leiomyomata: etiology, symptom-

atology, and management. Fertil Steril. 1981;36:433–445.

2. Serden SP, Brooks PG. Treatment of abnormal uterine bleeding with the

gynecologic resectoscope. J Reprod Med. 1991;36:697–699.

3. Lumsden MA. Modern management of fibroids. Obstet Gynaecol

Reprod Med. 2010;20:82–86.

4. Klatsky PC, Tran ND, Caughey AB, Fujimoto VY. Fibroids and repro-

ductive outcomes: a systematic literature review from conception to

delivery. Am J Obstet Gynecol. 2008;198:357–366.

5. Lefebvre G, Vilos G, Allaire C, et al. The management of uterine

leiomyomas. J Obstet Gynaecol Can. 2003;25:396–418.

6. Ubaldi F, Tournaye H, Camus M, et al. Fertility after hysteroscopic

myomectomy. Hum Reprod Update. 1995;1:81–90.

7. Polena V, Mergui JL, Perrot N, et al. Long-term results of hysteroscopic

myomectomy in 235 patients. Eur J Obstet Gynecol Reprod Biol. 2007;

130:232–237.

8. Jansen FW, Vredevoogd CB, van Ulzen K, et al. Complications of

hysteroscopy: a prospective, multicenter study. Obstet Gynecol. 2000;

96:266–270.

Peivandi et al. Uterine Cavity-Myoma Fistula after Hysteroscopic Myomectomy 537

9. Propst AM, Liberman RF, Harlow BL, Ginsburg ES. Complications of

hysteroscopic surgery: predicting patients at risk. Obstet Gynecol.

2000;96:517–520.

10. Kelly BA, Bright P, Mackenzie IZ. Does the surgical approach used for

myomectomy influence the morbidity in subsequent pregnancy?

J Obstet Gynaecol. 2008;28:77–81.

11. De Iaco PA, Muzzupapa G, Golfieri R, Ceccarini M, Roset B,

Baroncini S. A uterine wall defect after uterine artery embolization

for symptomatic myomas. Fertil Steril. 2002;77:176–178.

12. Sultana CJ, Goldberg J, Aizenman L, Chon JK. Vesicouterine fistula

after uterine artery embolization: a case report. Am J Obstet Gynecol.

2002;187:1726–1727.

13. Ogliari KS, Mohallem SV, Barrozo P, Viscimi F. A uterine-myoma

communication after uterine artery embolization: two case reports.

Fertil Steril. 2005;83:220–222.

14. Kim CK, Park BK.MR features of a fistula formation from the cavity of

a degenerated subserosal leiomyoma to the endocervical canal. Eur J

Radiol Extra. 2009;72:e83–e85.

15. Ng C, Lavery S, Hemingway A, Williamson R, et al. Successful spon-

taneous pregnancy following surgical removal of a post uterine artery

embolized necrotic fibroid capsule: a case report. Hum Reprod. 2006;

21:380–383.

16. De Iaco PA, Golfieri R, Ghi T, Muzzupapa G, Ceccarini M, Bovicelli L.

Uterine fistula induced by hysteroscopic resection of an embolized

migrated fibroid: a rare complication after embolization of uterine

fibroids. Fertil Steril. 2001;75:818–820.

17. Spies JB, Scialli AR, Jha RC, et al. Initial results from uterine fibroid

embolization for symptomatic leiomyomata. J Vasc Interv Radiol.

1999;10:1149–1157.

18. Goodwin SC, Walker WJ. Uterine artery embolization for the treat-

ment of uterine fibroids. Curr Opinion Obstet Gynecol. 1998;10:

315–320.

19. Borghi M, Lipowicz R, Baronio M, Carrascosa P, Mielnikowicz T,

Papier S. Ongoing pregnancy after a corrective surgery of an endomyo-

metrial fistula diagnosed by the use of virtual hysterosalpingography in

an infertile patient. Fertil Steril. 2008;90(suppl):S447.