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doi:10.1111/jog.12515

J. Obstet. Gynaecol. Res. Vol. 41, No. 2: 314–318, February 2015

Temporary balloon occlusion of the uterine arteries to control hemorrhage during hysterectomy in a case of uterine arteriovenous fistula Naoko Yamamoto1, Kaori Koga1, Masaaki Akahane2*, Osamu Wada-Hiraike1, Tomoyuki Fujii1 and Yutaka Osuga1 1

Department of Obstetrics and Gynecology, School of Medicine, 2Department of Radiology, The University of Tokyo, Tokyo, Japan

Abstract Arteriovenous fistula is any abnormal connection between an artery and a vein that bypasses the normal capillary bed and shunts arterial blood directly to the venous circulation. Uterine arteriovenous fistula (UAVF) is a potentially life-threatening condition by causing massive bleeding. This report describes a case of UAVF with massive hemorrhage. Prior to surgery, endovascular catheters for balloon occlusion were placed within bilateral uterine arteries. During surgery, the surgeon requested temporary balloon inflation for navigating and identifying inflow arteries. The balloon was kept inflated during the ligation of the vessels. Once ligation was completed, the balloon was deflated to confirm hemostasis. A total hysterectomy with removal of the UAVF was successfully achieved without significant blood loss. The fistula, in the resected specimen, was confirmed histologically with Elastica van Gieson staining. The preoperative placement of endovascular balloon-occlusion catheters should be considered when hysterectomy is planned where UAVF is located at the cardinal ligament. Key words: balloon occlusion, Elastica van Gieson stain, histology, hysterectomy, uterine arteriovenous fistula.

Introduction Arteriovenous fistula (AVF) is any abnormal connection between an artery and a vein that bypasses the normal capillary bed and shunts arterial blood directly to the venous circulation.1 Uterine arteriovenous fistula (UAVF) is AVF that has developed in the uterus. UAVF is a potentially life-threatening condition by causing massive vaginal bleeding.2 Despite its histological definition, UAVF has been variably described as uterine arteriovenous malformation, cirsoid aneurysm, arteriovenous aneurysm and cavernous hemangioma.2,3 UAVF are managed by either surgical therapies, mainly hysterectomy, or radiological interventions such as

uterine artery embolization.3 Histological diagnosis is usually not made because specimens are not available unless surgical resection is performed, and if performed, most fistulas are histologically unremarkable. Herein, we present a case of UAVF that was successfully managed by a combination therapy, hysterectomy with preoperative placement of endovascular balloonocclusion catheters, and the fistula was histologically confirmed in the resected specimen.

Case Report A 59-year-old woman, gravida 4, para 3, presented to a local hospital with sudden and massive uterine

Received: March 7 2014. Accepted: June 5 2014. Reprint request to: Dr Kaori Koga, Department of Obstetrics and Gynecology, The University of Tokyo, 7-3-1 Hongo, Bunkyo, Tokyo 113-8655, Japan. Email: [email protected] *Current affiliation: NTT Medical Center Tokyo.

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© 2014 The Authors Journal of Obstetrics and Gynaecology Research © 2014 Japan Society of Obstetrics and Gynecology

UAVF hysterectomy with balloon occlusion

bleeding. The patient had a history of normal vaginal deliveries at the age of 24, 27 and 32 years, and a dilation and curettage for terminating pregnancy at the age of 25 years. The woman reached menopause at the age of 56 years. An examination using a vaginal speculum revealed bleeding through the cervical canal, and the volume increased after attempting an endometrial biopsy. Tight vaginal packing with povidone-iodinesoaked, rolled gauze temporally controlled the bleeding, although the effect was not complete. The patient was transferred to our hospital for evaluation and management of uncontrolled uterine bleeding. Hemorrhaging aside, the patient did not present with abdominal pain or any other complaint. On examination with a speculum, a pulsating mass of 1 cm in diameter was observed beneath the vaginal mucosa and at the 9 o’clock position of the vaginal vault. This pulsating mass was visualized by transvaginal ultrasonography as a highly vascularized mass in the right parametrium. Magnetic resonance angiography (MRA) was performed for further analysis and it detected a vascular tangle, located at the right cardinal ligament, by the right uterine artery (Fig. 1); supplied by bilateral uterine arteries, ovarian arteries and vaginal arteries. Further, digital subtraction angiography detected early venous drainage during the arterial phase, suggesting direct connectivity between the artery and vein (Fig. 2),

although the fistula per se was not detected using this technique. Collectively, a diagnosis of UAVF with hemorrhage was made. A single uterine artery embolization was considered to be ineffective for this case given the large diameter and high velocity of blood flow at the lesion; we therefore decided to conduct a total abdominal hysterectomy with removal of the UAVF. Prior to the surgery, endovascular catheters for balloon occlusion were placed within bilateral uterine arteries in an attempt to reduce the risk of intraoperative, uncontrolled hemorrhage. Once confirmed that the balloon inflation was able to occlude blood flow (Fig. 3), the balloons were left deflated in order to avoid rapid development of collateral circulations. During surgery, the UAVF with massive hemorrhage into the uterine cavity was observed as a pulsating mass composed of tortuous arteries and veins on the right side of parametrium at the level of the cardinal ligament (Fig. 4). After dissection of round ligaments and infundibulopelvic ligaments, the anterior leaf of the broad ligament was dissected and the bladder was mobilized inferiorly. The vessels of UAVF were skeletonized by removing any overlying avascular areolar tissue. The ureter was identified, mobilized downward until the point where it crossed under the UAVF to

Figure 1 Original non-enhanced magnetic resonance angiography indicating the tortuous right uterine artery. rUA, right uterine artery; U, uterus.

Figure 2 Digital subtraction angiography of the right uterine artery indicates an enlarged vascular mass and early draining vein. V, the vein.

© 2014 The Authors Journal of Obstetrics and Gynaecology Research © 2014 Japan Society of Obstetrics and Gynecology

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Figure 4 Intraoperative findings of the uterine arteriovenous fistula (UAVF). The UAVF was observed as a pulsating mass composed of tortuous arteries and veins on the right side of the parametrium at the level of the cardinal ligament. Fundus, uterine fundus; Lt, left side; Rt, right side. Figure 3 Photograph during angiography which was taken when balloons in bilateral uterine arteries were inflated. Note that the contrast media remained in the uterine arteriovenous fistula and did not flow to the draining vein or other areas.

ensure the ligation of UAVF might not cause the ureteral injury. The surgeon requested temporary balloon inflation for navigating and identifying inflow arteries. The balloon was kept inflated during the ligation of vessels. Once ligation was completed, the balloon was deflated to confirm hemostasis. A total hysterectomy with removal of the UAVF was successfully achieved with an operative time 3 h and 12 min, blood loss of 700 mL, and a blood transfusion was not required. The weight of the resected uterus was 166 g. The balloonocclusion catheters were removed after surgery without complications. The patient’s postoperative course was unremarkable. Histological examination revealed an artery directly connected to a vein, which is consistent with the diagnosis of AVF (Fig. 5). Recurrence was not detected up to 2 years after surgery.

Discussion This is a case report of UAVF managed by a combination therapy, hysterectomy with preoperative place-

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Figure 5 Histological findings of the lesion (Elastic van Gieson staining). Note the transition from the arterial to the venous wall; the arterial media and the internal elastic lamina have disappeared. This finding is consistent with the diagnosis of arteriovenous fistula. A, artery; V, vein. Bar = 2 mm.

ment of endovascular balloon-occlusion catheters. This is also the first published case of UAVF that reports the presence of arteriovenous continuity in the resected specimen.

© 2014 The Authors Journal of Obstetrics and Gynaecology Research © 2014 Japan Society of Obstetrics and Gynecology

UAVF hysterectomy with balloon occlusion

Regarding the management of UAVF, hysterectomy was the treatment of choice, before transcatheter embolization procedures were widely introduced and accepted.4 Even today, hysterectomy is the first choice for management of UAVF, especially for cases where women do not need to preserve reproductive function, because single uterine artery embolization is not always successful and failure of embolization may result in repetitive bleeding that requires repeated embolizations or a hysterectomy.2,5,6 Hysterectomy, however, can lead to massive blood loss,4,7 and may require invasive surgeries such as radical hysterectomy.7 In the present case, hysterectomy was chosen over embolization because the patient had no need of uterine preservation, and a single uterine artery embolization was considered to be ineffective due to the large diameter and the high velocity of blood flow at the lesion. Alternatively, we introduced combination therapy, hysterectomy with preoperative placement of endovascular balloon-occlusion catheters, which facilitates temporary occlusion of the uterine arteries to control hemorrhage during the surgery. Preoperative placement of endovascular balloonocclusion catheters for pelvic surgery has been developed with the aim of decreasing intraoperative blood loss, by means of temporary control of pelvic circulation, primarily by occluding the internal iliac arteries or their branches. This technique was first introduced for cesarean section or cesarean hysterectomy for cases of abnormal placentation such as placenta previa and placenta increta.8–10 Over the past 15 years, this technique has been used more frequently to treat patients at risk of obstetric hemorrhage. Indeed, several studies indicate that preoperative placements of balloon-occlusion catheters were associated with reduced intraoperative blood loss and transfusions.11,12 On the other hand, using this technique in gynecologic disease is less common; its efficacy has only been reported in limited conditions such as hysterectomy for cervical myoma.13 Applying this strategy to hysterectomy to treat UAVF was first reported by Soeda and colleagues,14 where they placed a catheter with occlusion balloon in the iliac artery and left the balloon inflated during the hysterectomy. The present case is the second report with slight modifications; we placed the occlusion balloon in the uterine artery and inflated the balloon only when needed. This approach has the following advantages: inflating and deflating occlusion balloons helps the surgeon navigate feeding and draining vessels within the vascular tangles, and occlusion of the blood supply during vessel ligations allows for better visualization.

Consequently, favorable surgical outcomes were obtained in the present case, although the significance of these advantages is subjective and may be smaller than when this strategy is applied for more challenging cases such as larger uterus or cervical mass. Further studies, such as randomized controlled studies, are warranted to investigate the efficacy of this procedure. To the best of our knowledge, this is the first report of UAVF where the fistula was histologically confirmed in the resected specimen. Histological diagnosis is usually not made because specimens are not available unless surgical resection is performed, and if performed, most fistulas are histologically unremarkable.2 In the present case, Elastica van Gieson staining clearly shows the transition from the arterial to the venous wall (fistula), where the arterial media and the internal elastic lamina have disappeared. This finding also revealed that the size of the fistula was as small as 2 mm, which explains why the preoperative MRA did not detect the fistula per se. In summary, a case of histologically confirmed UAVF, which was successfully managed by temporary balloon occlusion of the uterine arteries during hysterectomy, is presented. We propose that preoperative placement of an endovascular balloon-occlusion catheter should be considered when hysterectomy is planned in cases where a UAVF is located at the cardinal ligament.

Acknowledgments The authors thank Department of Pathology in the University of Tokyo for histological reviewing, Dr Masanori Maruyama for recruitment of the patient, and Dr Kate Hale for editing the manuscript.

Disclosure The authors have nothing to disclose.

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4. Hoffman MK, Meilstrup JW, Shackelford DP, Kaminski PF. Arteriovenous malformations of the uterus: An uncommon cause of vaginal bleeding. Obstet Gynecol Surv 1997; 52: 736– 740. 5. Ghai S, Rajan DK, Asch MR, Muradali D, Simons ME, TerBrugge KG. Efficacy of embolization in traumatic uterine vascular malformations. J Vasc Interv Radiol 2003; 14: 1401– 1408. 6. Hasegawa A, Sasaki H, Wada-Hiraike O et al. Uterine arteriovenous fistula treated with repetitive transcatheter embolization: Case report. J Minim Invasive Gynecol 2012; 19: 780–784. 7. Moulder JK, Garrett LA, Salazar GM, Goodman A. The role of radical surgery in the management of acquired uterine arteriovenous malformation. Case Rep Oncol. 2013; 6: 303–310. 8. Hudon L, Belfort MA, Broome DR. Diagnosis and management of placenta percreta: A review. Obstet Gynecol Surv 1998; 53: 509–517. 9. Weeks SM, Stroud TH, Sandhu J, Mauro MA, Jaques PF. Temporary balloon occlusion of the internal iliac arteries for control of hemorrhage during cesarean hysterectomy in a patient with placenta previa and placenta increta. J Vasc Interv Radiol 2000; 11: 622–624.

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10. Kidney DD, Nguyen AM, Ahdoot D, Bickmore D, Deutsch LS, Majors C. Prophylactic perioperative hypogastric artery balloon occlusion in abnormal placentation. AJR Am J Roentgenol 2001; 176: 1521–1524. 11. Sadashivaiah J, Wilson R, Thein A, McLure H, Hammond CJ, Lyons G. Role of prophylactic uterine artery balloon catheters in the management of women with suspected placenta accreta. Int J Obstet Anesth 2011; 20: 282–287. 12. Tan CH, Tay KH, Sheah K et al. Perioperative endovascular internal iliac artery occlusion balloon placement in management of placenta accreta. AJR Am J Roentgenol 2007; 189: 1158– 1163. 13. Takeda A, Koyama K, Imoto S, Mori M, Nakano T, Nakamura H. Temporary endovascular balloon occlusion of the bilateral internal iliac arteries to control hemorrhage during laparoscopic-assisted vaginal hysterectomy for cervical myoma. Eur J Obstet Gynecol Reprod Biol 2011; 158: 319– 324. 14. Soeda S, Ushijima J, Furukawa S et al. Uterine arteriovenous malformation formed in a large uterine cervical myoma. Tohoku J Exp Med 2012; 228: 181–187.

© 2014 The Authors Journal of Obstetrics and Gynaecology Research © 2014 Japan Society of Obstetrics and Gynecology

Temporary balloon occlusion of the uterine arteries to control hemorrhage during hysterectomy in a case of uterine arteriovenous fistula.

Arteriovenous fistula is any abnormal connection between an artery and a vein that bypasses the normal capillary bed and shunts arterial blood directl...
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