Successful Management of Spontaneous Iliac Vein Rupture with Double Bare Stent Technique: A Case Report and Review of Literature

Abstract

Spontaneous rupture of the iliac vein is a rare cause of retroperitoneal hematoma. A misdiagnosis may delay the treatment and consequently put the patient in a life-threatening emergency. We report the case of a 73-year-old woman who presented with hemorrhagic shock from bleeding caused by a large left retroperitoneal hematoma. She was successfully treated with an endovascular approach by using a double bare stent technique. An extensive review of the literature was conducted and a total 44 articles with 50 patients were identified. Among these patients, 88.2% were women, 94.1% presented with a left-sided rupture, and the mortality rate was 13.7%. Spontaneous iliac vein rupture was more likely to occur in the left side in female patients. Conservative treatment was an option in hemodynamically stable patients. Exploratory laparotomy and surgical iliac vein repair was necessitated in most patients. Endovascular treatment including placement of covered stent and coil embolization had been widely used to treat spontaneous vein rupture since 2003. Double bare stent technique was also an effective alternative if a suitable covered stent was unavailable.

Keywords

case report iliac vein rupture interventional radiology vascular surgery

Introduction

Spontaneous iliac vein rupture (SIVR) is a rare and under-recognized clinical emergency. Patients may present with a retroperitoneal hemorrhage that is often misdiagnosed. Urgent exploratory laparotomy was often performed in these patients, but with the growing experience in the fields of endovascular technique, more and more endovascular repair has been used in the past 2 decades. Covered stents were commonly deployed across the rupture site to seal the leakage. However, a suitable size is not always available, especially during an emergency setting.

In this study, we report an elderly woman who presented with a large retroperitoneal hematoma and hemorrhagic shock caused by SIVR. The presence of DVT proximal to the rupture was also identified. Our report details the successful treatment of SIVR with double bare stent technique. The patient has agreed with the publication of this case report and written consent has been obtained.

Case Report

A 73-year-old female was admitted to our emergency department due to severe abdominal pain persisted for 10 hours and accompanied with transient loss of consciousness. She was hemodynamically unstable upon arrival. Her vital signs were 59/32 mmHg blood pressure, and 139 beats/min pulse rate. The physical examination was significant for a tender, non-pulsatile abdominal mass (approximately 10-cm x 15-cm in size) at the left lower quadrant and a swollen left lower limb with increased tension. Her left dorsalis pedis and posterior tibial artery pulses were palpable. She has a past medical history of hypertension for 10 years which was poorly controlled. Biochemical results revealed an elevated white blood cell count (15.39 × 10⁹/L), low platelet count (70 × 10⁹/L), low hemoglobin level (64 g/L), elevated D-dimer (24.2 mg/L), and prolonged activated partial thromboplastin time (APTT) (53.20s). Her computed tomography (CT) report revealed a retroperitoneal hematoma measuring 14.8-cm × 6.3-cm, along with fluid accumulation in the abdominal cavity.

We immediately initiated resuscitation with crystalloid and blood transfusion to manage her hemorrhagic shock. She was transferred to the intensive care unit (ICU) once her blood pressure has reached 86/55 mmHg. A contrast-enhanced computed tomography was arranged, which revealed a large retroperitoneal hematoma measuring 4.6-cm × 11.1-cm × 19.2-cm and accumulation of blood and fluid in the abdominopelvic cavity. Suspected left iliac vein thrombosis was also noted on the CT scan (Figure 1).

Figure 1.

Abdominal CT revealed a retroperitoneal hematoma occupying the space from below the left kidney to the entire pelvic cavity.

An urgent angiogram and venogram were performed to identify the source of bleeding. Aortoiliac angiography was performed using left femoral access. No contrast extravasation was revealed. A small aneurysm was found on the distal end of a lumbar artery branch, no contrast leakage identified. It was embolized with 2 3-mm × 3.3-mm coils (VortX Diamond-18, Boston Scientific, USA).

Venogram using right femoral access was performed. A .035^″ guidewire was advanced to the left iliac vein with the support of a 5F Cobra catheter (Cordis, USA), thrombosis of the left common iliac and external iliac veins was visualized, and significant contrast extravasation was found within the left external iliac vein (Figure 2(a)). Left external iliac vein rupture was confirmed. The external iliac vein was measured 10 mm in diameter, there was no suitable covered stent available. Therefore, we attempted to place 2 overlapped bare stent to cover the rupture.

Figure 2.

Angiography revealed significant contrast extravasation from the left external iliac vein (A). Angiography performed after covering the rupture site with double bare stent technique revealed a patent left common iliac vein without contrast extravasation (B).

Left iliac vein compression syndrome (VCS) was also suspected since resistance was met during advance of the guidewire and catheter. Venoplasty of the left common iliac vein was performed using a 10-mm x 80-mm balloon (Ultraverse, Bard, USA), stenosis was seen on fluoroscopy during venoplasty which was consistent with VCS. A 12-mm × 90-mm stent (Wallstent, Boston Scientific, USA) was placed at the proximal segment of the left iliac vein to cover the rupture site. Another 12-mm × 60-mm stent (Wallstent, Boston Scientific, USA) was placed to overlap the rupture site. The length of overlap coverage was approximately 5-cm. There was no contrast extravasation from the left iliac vein on venogram (Figure 2(b)). After placement of an inferior vena cava filter (Option Elite, Argon, USA), pharmacomechanical thrombectomy (PMT) was then performed using a 6F catheter (AngioJet, Boston Scientific, USA) Completion venography results showed the left iliac vein was patent without filling defect or contrast extravasation.

Anticoagulation (Nadroparin 4000U q12H) and blood transfusion were administered postoperatively. Her postoperative blood pressure was within the normal range. On the third postoperative day, her hemoglobin level has raised to 91 g/L. Physical examination found an obvious reduction of tension in her left lower limb. The circumferences of her thigh (15-cm above the knee) and calf (10-cm below the knee) have reduced by 4.1-cm and 2.0-cm respectively. Postoperative duplex ultrasound showed her left iliac, femoral, and popliteal veins were patent. She was discharged and prescribed with oral rivaroxaban 20 mg QD.

Follow up CT at 1 and 3 months after the procedure showed gradual size reduction of the retroperitoneal hematoma (Figure 3). USG of the lower limb showed patency of the stent and venous system of the left lower limb. She had successful IVC filter retrieved 3 months after the operation.

Figure 3.

CT scan results from the same segments performed upon admission (A), 1-month (B), and 3-month (C) post operation reveals gradual size reduction of the retroperitoneal hematoma.

Discussion

The first case of SIVR was reported by Hossne WS et al.¹ in 1961. An extensive review of the literature was conducted and a total 44 articles with 50 patients were identified. The characteristics of the included studies had been summarized in Table 1. Most of the published literature are case reports. Among these patients, 88.2% were women, 94.1% presented with a left-sided rupture, and the mortality rate was 13.7%.

Table 1.

Summary of characteristics of the included studies on spontaneous iliac vein rupture.

Author, year	Country	Age/Gender	Predisposing Factor (s)	Clinical finding(s)	Rupture Site	Concomitant IVT	Iliac Vein Compression	SIVR Treatment	Outcome
Hossne,¹ 1961	Brazil	48/F	Constipation	LAP, LOC	REIV	+		Ligation	Survived
Herrin,² 1975	USA	67/F	Fall	LAP, LLES	LCIV	+		OAR	Survived
Brown,³ 1977	USA	75/F	Fall	LAP, LOC	LCIV	+		OAR	Survived
Miles,⁴ 1977	Canada	70/F	—	LBP	LCIV			OAR	Survived
McDonald,⁵ 1980	USA	46/F	Thrombophlebitis	AP, chest pain	LEIV	+	+	Ligation	Died
Elliot,⁶ 1982	UK	72/F	Bending	LAP	REIV			OAR	Survived
Noszczyk,⁷ 1983	Poland	48/M	Bending, pneumonia	LAP	LEIV	+	+	OAR	Survived
Stock,⁸ 1986	UK	79/F	Vein compression	LAP, LLES	LCIV	+	+	OAR	Survived
Forsberg,⁹ 1988	Sweden	83/F	Sigmoiditis, diarrhea	LAP	LEIV	−		OAR	Survived
Forsberg,⁹ 1988	Sweden	74/F	Defecation	LFP, LLES	LEIV	+		OAR	Survived
Copeland,¹⁰ 1989	UK	63/F	Hypertension	LIFP	LCIV	−		OAR	Survived
Hill,¹¹ 1990	UK	52/F	Defecation	LHP	LCIV	−	−	OAR	Died
Van Damme,¹² 1993	Belgium	63/M	Defecation, sigmoid cancer	LAP	LCIV	+	+	OAR	Survived
Majeed,¹³ 1993	UK	49/F	Bending	LAP	LEIV	+		OAR	Survived
Yamada,¹⁴ 1995	Japan	66/F	—	LAP, LLES	LEIV	+	+	OAR	Survived
Plate,¹⁵ 1995	Sweden	78/F	—	LLAP, LLES, LLEP	LEIV	+		OAR	Survived
Lin,¹⁶ 1996	China	69/F	Bending	LAP	LEIV	+		OAR	Survived
DePass,¹⁷ 1998	Canada	68/F	Bowling	LLAP	LCIV			OAR	Survived
Graschignard,¹⁸ 2000	France	58/F	Previous partial hysterectomy	LIFP	LC-EIV	+	−	OAR + Ligation	Survived
Pedley,¹⁹ 2002	UK	66/F	—	LLAP, LLES, LLEP	LCIV	+		Ligation	Survived
Jayazeri,²⁰ 2002	France	45/F	Bending	LLES, LLEP	LEIV	−		OAR	Survived
Cho,²¹ 2003	Korea	58/F	—	LIFP	LCIV	+		Conservative	Survived
Kwon,²² 2004	Korea	62/F	Hypertension, Constipation	LAP	LEIV	−	+	OAR	Survived
Zieber,²³ 2004	USA	48/F	DVT	LBP, LLES, LLEP	LEIV	+		Double bare stent	Survived
Yasuga,²⁴ 2004	Japan	79/F	—	LAP	LEIV	+		OAR	Survived
Tannous,²⁵ 2006	USA	56/M	DVT, IVC filter	LAP	LEIV	−		OAR	Died
Kim,²⁶ 2007	Korea	60/F	—	LLAP, LLES	LCIV	+	+	OAR	Survived
Jiang,²⁷ 2010 (4 cases)	China	46-70/F	Defecation, bending, walking	LLP, LLAP, LLES, LLEP	LCIV (3), LEIV (1)	4/4	3/4	OAR (3), Ligation + Palma-Dale bypass graft (1)	3 survived, 1 died
Zang,²⁸ 2011	China	62/F	Fall, defecation	LAP, right lower limb immobility	REIV	+		OSR	Survived
Deng,²⁹ 2011	China	63/F	—	LAP, LLEP	LCIV	+		OAR	Survived
Kim,³⁰ 2011	Korea	72/F	Obesity, sudden position change	LAP, BP	LEIV			OAR	Survived
Chen,³¹ 2013	China	51/F	—	LLAP, LOC, LLES	LCIV	+		OAR	Survived
Hughes,³² 2013	USA	49/F	Thrombophlebitis, perimenopause	LLES, LLEP, erythema	LCIV	+	+	Thrombolysis + IVC filter placement	Died
Sadaghianloo,³³ 2014	France	73/M	—	LLES	LEIV	+	+	Conservative then OSR	Survived
Kim,³⁴ 2015	Korea	62/F	—	LLES	LEIV	+	−	Conservative	Survived
Hosn,³⁵ 2016	USA	39/F	—	LLP, LOC, PCD	LCIV (distal)	+	+	OAR + stenting	Survived
Joseph,³⁶ 2017	USA	51/F	Uterine fibroids	LLP, LAP, LLES, LLEP	LC-EIV	−	−	OAR	Died
Chen,³⁷ 2018	China	71/F	—	LAP, LOC, LLES	LEIV			Cover stent	Survived
Ingram,³⁸ 2019	USA	58/F	Exercise	LAP, LLES, erythema	LIV	+	+	Conservative, then mechanical thrombectomy + stenting	Survived
Molière,³⁹ 2020	France	70/F	—	LIFP, LLES	LIV	+	+	Conservative	Survived
Agilinko,⁴⁰ 2020	UK	60/F	—	LLEP, LLES	LCIV	+	+	OAR	Died
McCready,⁴¹ 2021	USA	76/M	Exercise	LOC, PCD	LEIV	+	−	Amplatzer plugs + Azur coils	Survived
McCready,⁴¹ 2021	USA	77/M	DVT, hernia repair	LLAP, LLES	LEIV branch	+	+	IVC filter placement then OSR	Survived
Sueyoshi,⁴² 2021	Japan	82/F	Hypertension, constipation	LOC, LLAP, LBP	LEIV	+		Embolization (NBCA/Lipiodol mixture)	Survived
Nishimoto,⁴³ 2021	Japan	69/F	—	LBP, LLES	LEIV	+	+	Conservative, then Forgarty venous thrombectomy + cover stent	Survived
Zheng,⁴⁴ 2021	China	65/F	—	LLAP, LLES	LEIV	+	+	Cover stent	Survived
Zheng,⁴⁴ 2021	China	53/F	—	AP, LBP	LEIV	+	+	Cover stent	Survived
Qi, 2022 (Present report)	China	73/F	—	LLAP, LLES	LEIV	+	+	Double bare stent	Survived

AP, abdominal pain; IVC, inferior vena cava; LAP, lower abdominal pain; LLAP, left lower abdominal pain; LBP, lower back pain; LFP, left flank pain; LIFP, left iliac fossa pain; LLES, left lower extremity swelling; LLEP, left lower extremity pain; LCIV, left common iliac vein; LEIV, left external iliac vein; REIV, right external iliac vein; OAR, open abdomen repair; PCD, phlegmasia cerulia dolens; SIVR, spontaneous iliac vein rupture.

The exact etiology of the disease is still unknown, but various theories have been suggested, namely the increased fragility of vessel walls and increased venous pressure in the iliac veins. Estrogen is known to promote vessel compliance, and postmenopausal women deplete in them, consequently resulting in a loss of such protective effect, and causing the weakening of the venous wall.²⁵ Chronic inflammation and venous thrombophlebitis can potentially cause the venous wall to lose its elasticity.⁴⁵ In our literature review, we found that 75% of patients had a concomitant venous thromboembolism. Many of the patients without concomitant DVT might have proximal venous obstruction. Absent venous hypertension, it seems highly unlikely that such rupture could occur, as intra-abdominal pressure usually approximates or exceeds normal venous pressures and would therefore tamponade any tendency for bleeding or hematoma expansion. Currently, we are una ble to determine whether the thrombus or rupture existed first. However, thrombophlebitis remains 1 of the potential causative factors that cannot be ignored. Zhang et al.⁴⁶ reported that 62.5% of patients have displayed such histopathological evidence. Some studies described that the iliac vein was already too weak and beyond repair when surgeons found its defect during exploratory laparotomy.^18,19 May-Thurner syndrome is a condition leading to venous outflow obstruction due to extrinsic venous compression in the iliocaval venous territory. This makes it 1 of the common causes of venous hypertension.³⁸ The syndrome was especially recognized and identified in the case studies published in the recent 10 years (2010-2021). In this group, 75% (18/24) were reported to have concomitant iliac vein compression. Additionally, certain actions that may suddenly increase venous pressure, such as hurdling, squatting, strenuous exercise, and defecation, are common triggers of iliac vein rupture.

Spontaneous iliac vein rupture often presents as persistent pain in the lower left abdomen, accompanied with hypotension. Patients who experience massive bleeding may present with syncope first. In most cases, patients have an underlying deep vein thrombosis (DVT) that causes swelling of the lower limbs. In our review, we found that 50% (25/50) of the patients presented with left lower limb swelling. Tannous et al.²⁵ found in their review of 33 cases, 79% had a concomitant venous thromboembolism. CT scan and MRI are useful imaging modalities in the diagnosis of iliac vein rupture. In most of the cases, retroperitoneal hematoma could be identified on CT scan or MRI. Contrast extravasation can be seen on CT scan if there is active bleeding. However, it is difficult to differentiate venous rupture from arterial bleeding. Duplex ultrasound has a low sensitivity in diagnosis of iliac vein rupture. Venogram is the golden standard for diagnosis of iliac vein rupture. However, it is invasive and may not be immediate available in the emergent setting.

Conservative treatment is an option that is only applicable to hemodynamically stable patients.^21,34,38,39 Traditional laparotomy and surgical iliac vein repair is the most commonly used open surgical approach. Endovascular treatment of SIVR was first reported in 2003. Since then, a large variety of devices including covered stent, with or without embolization coils and glue have been used. The bare stent technique for treatment of SVIR has been reported by Zieber et al.²³ previously. It is effective to stop bleeding by cover the rupture site, decreasing venous pressure, restoration of venous blood flow and allowing the hematoma surrounding the vein to tamponade further bleeding. In this patient, we used 2 overlapped bare stents instead of a covered stent beacause a 13-mm diameter covered stent was not immediately available. According to a multi-institutional international practice pattern survey, most common stent diameters that used for endovascular iliocaval reconstruction were 14-mm in the common iliac vein, and 12-mm in the external iliac vein.⁴⁷ We used 2 12-mm diameter overlapped Wallstents in this case, landing from common iliac vein to external iliac vein. The size was suitable for external iliac vein. However, a 12 mm diameter stent might be a little undersized for common iliac vein. In this patient, there was suspected left iliac vein compression during venoplasty. Therefore, we chose to use 2 12-mm diameter stents. The first Wallstent was deployed at left common iliac vein through left femoral vein access. The proximal end of the second Wallstent was 2-3 cm proximal to the rupture site. There were overlapped stents covering the rupture site at least 2 cm proximally and 2 cm distally. We deployed 2 overlapped stents to cover the rupture and subsequently performed PMT to remove the thrombosis. As there was still contrast extravasation on venogram, control of the bleeding was the first priority in the emergency setting. PMT would increase the risk of bleeding if it was done before iliac vein stenting. The stent was patent after thrombectomy in this patient.

Conclusion

Spontaneous rupture of the iliac vein is a rare but life-threatening condition. Prompt diagnosis and early resuscitation are essential. Traditional laparotomy and surgical iliac vein repair is the most commonly used open surgical approach. Minimally invasive endovascular treatment has gained popularity in the past 2 decades with a satisfactory outcome. In selected cases, endovascular approach with double bare stent placement is an effective treatment option.

Footnotes

Author Contribution

Conceptualization: Hao-Shan QI

Data curation: Hao-Shan QI

Formal analysis: Hai-Lei LI

Investigation: Hao-Shan QI, Kristine J.S. KWAN

Methodology: Hai-Lei LI

Project administration: Hai-Lei LI

Software: Hao-Shan QI, Hai-Lei LI, Kristine J.S. KWAN

Supervision: Hai-Lei LI

Validation: Hai-Lei LI, Kristine J.S. KWAN

Visualization: Hao-Shan QI, Kristine J.S. KWAN

Writing – original draft: Hao-Shan QI

Writing – review & editing: Hai-Lei LI, Kristine J.S. KWAN

Declaration of conflicting interests

The author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.

Funding

The author(s) received no financial support for the research, authorship, and/or publication of this article.

Informed Consent

The patient has agreed with and consented for the publication of their case.

ORCID iDs

Hao-Shan QI

Kristine J.S. KWAN

Hai-Lei LI

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