Spontaneous Rupture of the Right External Iliac Vein Hong Zang,1 Ting Zhang,2 Ai-Lian Liu,2 Feng Wang,3 and Hong Shi,1 Liaoning, People’s Republic of China
Spontaneous rupture of an iliac vein is a very rare condition, with 34 reported cases in the previously published data and only two cases involving the right side. We report the third case of spontaneous rupture of the right external iliac vein. A 62-year-old woman presented with sudden onset of lower abdominal pain and an inability to move the right lower extremity shortly after stretching her right leg backward. A contrast computed tomography demonstrated a massive pelvic retroperitoneal hematoma and a thrombus extending from the inferior vena cava to the right iliac veins. An inferior vena cave filter was inserted using interventional radiology, followed by an exploratory laparotomy. A 1.5-cm laceration in the right external iliac vein was uncovered and repaired. The etiology, clinical features, and treatment of spontaneous iliac vein rupture are discussed.
Spontaneous rupture of an iliac vein is a very rare condition. The 32 reported cases occurring in the left-sided iliac veins were associated with Maye Thurner syndrome (i.e., the compression of the left common iliac vein by the overlying right common iliac artery).1,2 There have been only two reported cases involving the right side.3,4 Our patient is the third case of spontaneous rupture of the right external iliac vein to be reported.
CASE REPORT Our case was a 62-year-old healthy woman who presented with sudden onset of lower abdominal pain and an inability to move the right lower limb. 1 Department of Obstetrics and Gynecology, The First Affiliated Hospital, Dalian Medical University, Liaoning, People’s Republic of China. 2
Department of Radiology, The First Affiliated Hospital, Dalian Medical University, Liaoning, People’s Republic of China. 3 Department of Interventional Radiology, The First Affiliated Hospital, Dalian Medical University, Liaoning, People’s Republic of China.
Correspondence to: Hong Shi, MD, PhD, Department of Obstetrics and Gynecology, The First Affiliated Hospital, Dalian Medical University, 116001 Dalian, Liaoning, People’s Republic of China, E-mail:
[email protected] Ann Vasc Surg 2011; 25: 701.e1-701.e4 DOI: 10.1016/j.avsg.2011.02.006 Ó Annals of Vascular Surgery Inc. Published online: April 21, 2011
The patient had the acute onset of severe lower abdominal pain when at 9 A.M. she stepped down from a 100-cmhigh table in the backward direction using her right leg. After walking a few steps her right lower limb became immobile, and she fell. Thereafter, she defecated but presented with urination inability. Laboratory examinations in a local hospital at 11 A.M. showed anemia, with hemoglobin (HB) of 95 g/L and hematocrit of 31.5%, and hyperglycemia, with an increased level of blood glucose by 684 mg/dL. Abdominal ultrasonography and noncontrast pelvic computed tomography (CT) both discovered an irregular and heterogeneous soft-tissue mass measuring 80 37 cm2 surrounding the bladder. The woman was transported to the emergency room of the First Affiliated Hospital of Dalian Medical University at 7 P.M. in an ambulance because of hyperglycemia and the unknown cause of the abdominal mass. On arrival, the patient was administered an insulin infusion and indwelling urethral catheterization. Her blood pressure was 90/57 mm Hg and pulse rate was 110/minute. Abdominal palpation detected a large, tender, nonpulsatile mass in the right lower quadrant. The lower right limb could passively be mobile and was not swollen. Blood tests revealed white blood cell count of 15.4 109/L, HB of 85 g/L, and platelet count of 127 109/L. Coagulation time was within normal limits, but fibrinogen level was lower by 1.11 g/L. A noncontrast CT of abdomen and pelvis revealed a pelvic mass measuring 10 5.4 cm2 and partly filled with blood. The patient was admitted to a gynecologic ward with the diagnosis of a hemorrhagic mass in the pelvic cavity. Vital signs were stable overnight. The insulin infusion was continued to decrease her blood glucose level.
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Fig. 1. Computed tomographic image displaying a pelvic hematoma measuring 10 5.4 cm2 (arrows).
Fig. 3. The filter (arrows) was implanted at the inferior vena cava using interventional radiology.
Fig. 2. Computed tomographic image revealing a large retroperitoneal hematoma extending to level of the right kidney (small arrows) and dilation of the inferior cava vein with a thrombus (big arrow).
Further investigations were performed on the next day. Hepatic and renal functions were normal. Blood glucose level was reduced to 178 mg/dL. Blood tests in series showed a gradual decline in levels of white blood cell count (16.44-9.52-9.03 109/L), HB (85-64.2-62.2 g/ L), and platelet count (127-64-62 109/L). Moreover, there was a prolongation of the activated partial thromboplastin time (32.5-37.7 seconds) and an increased D-dimer level (851-1,663 mg/L) accompanied by a sustained low level of fibrinogen (1.11-1.12 g/L). The changes indicated early development of disseminated intravascular coagulation. A contrast CT evaluation showed that besides the pelvic hematoma (Fig. 1), a massive retroperitoneal hematoma was present up to the level of the right kidney (Fig. 2). Furthermore, a thrombus was demonstrated extending from the inferior vena cava at the level of the
renal hilum (Fig. 2) to the right common, external, and internal iliac veins. In addition, a culdocentesis was negative. The results were not supportive of a gynecological disorder. Therefore, the patient was presented to a group consultation of physicians in general surgery, hematology, interventional radiology, endocrinology, and urology. However, the source of hemorrhage could still not be determined. Thirty-four hours after the initiation of the condition, a vena cava filter was implanted at the inferior vena cava above the level of the renal vein from the right jugular vein using interventional radiology (Fig. 3) to prevent pulmonary and cerebral embolism. Shortly after, an exploratory laparotomy was performed. The uterus, ovary, and fallopian tubes were normal. The ‘‘pelvic hematoma’’ visualized by CT scan was located in the anterior abdominal wall surrounding the bladder between the peritoneum and muscle. The large hematoma was indeed on the right side of the retroperitoneum. Urology, general, and vascular surgeons were consulted successively. After evacuation of the clots, a 1.5-cm smooth rupture, with slow active bleeding, was uncovered in the posterolateral wall of the right external iliac vein with a fresh thrombus inside the lumen. The thrombus around the rupture was removed and the defect was sutured with a 5-0 prolene suture. The patient was treated with anticoagulation and insulin after the surgery. Type-2 diabetes mellitus was confirmed by the patient’s history of classic symptoms of polydipsia, polyphagia, polyuria, and hyperglycemia. The right lower limb became slightly swollen after the third day of the surgery. A color ultrasonography demonstrated thrombosis in the femoral and popliteal veins.
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Otherwise, the postoperative course was uneventful. The limb swelling gradually diminished, and blood glucose reached a satisfactory level. The patient was discharged after 11 days of hospitalization.
DISCUSSION The etiology of spontaneous rupture of an iliac vein remains ill-defined. Various factors have been suggested to be associated with its occurrence. The potential weakness of the venous wall may play an important role. Indeed, 85% of the reported cases (28 of 33) occurred in postmenopausal women with an average age of 60 years.1 Estrogen is known to exert a protective effect on compliance of blood vessels. Moreover, 79% of the reports (26 of 33) have documented clinical or histological evidence of venous thrombophlebitis, indicative of impaired elasticity of blood vessel by inflammatory factors.1 Because the condition occurs predominantly in the left iliac vein, it has been attributed to the compression of the left common iliac vein by the overlying right common iliac artery, so called MayeThurner syndrome.5 Without a history of trauma, bending/coughing or defecating, which raises the pressure of the left iliac vein between the right common iliac artery and inguinal ligament, is thought to be a predisposing factor to precipitate the development of the condition.1 There have been only two cases reported involving the right iliac vein.3,4 The first case was described in the Portuguese literature in 1961.3,6 A 48-year-old white Brazilian housewife presented abruptly with severe pain localized to the lumbar region after 2 days of malaise. Physical examination disclosed localized tenderness over the right lower quadrant. The location of the rupture of the iliac vein was not specified but was presumably on the right side.3,6 The second case was reported by Elliot and Ware in 1982.4 A 72-year-old Caucasian woman presented with severe lower abdominal pain, which had started suddenly while bending to open a cupboard. Abdominal examination revealed a tender swelling in the right iliac fossa. At laparotomy, a linear tear was revealed in the right external iliac vein which was repaired with a continuous suture.4 In our case, the third case of spontaneous rupture of the right external iliac vein, the patient presented with low abdominal pain and an inability to move the right lower extremity and urinate shortly after stretching the right leg backward. Abdominal examination disclosed a large tender, nonpulsatile mass in the right lower quadrant. The clinical features in our case were similar
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to those in the previously reported cases3,4 but different from those in the report of the left iliac vein rupture, in which a tender, nonpulsatile mass was located in the left lower quadrant.7 A unique feature of our case was that the patient was unable to move the right leg and urinate. These symptoms might be the result of pain originating in the abdominal and retroperitoneal hematoma. The rupture occurred on the right but not the left iliac vein, and this could be related to a special posture (i.e., extending the right leg backward) that raises the pressure of the right iliac vein. Contrast CT demonstrated in our case the pelvic and massive retroperitoneal hematoma as well as the extensive thrombus from the inferior vena cava to the right iliac veins. Therefore, CT examination would be helpful in making the diagnosis of a spontaneous rupture of the iliac vein.7 However, because of the rarity of the condition, we did not take it into consideration preoperatively. The rupture of the right external iliac vein was uncovered and sutured at the exploratory laparotomy. The treatment was successful but deep vein thrombosis was a postoperative complication, as reported in most of the previous cases in the literature.1 Therefore, the ideal course of treatment should consist of immediate cessation of bleeding and maintaining continuity in the deep-lying venous network.1 In summary, spontaneous iliac vein rupture is an uncommon vascular emergency. The condition should be considered in the differential diagnosis of abdominal hemorrhage in clinical practice, especially for postmenopausal women. It is more often affecting the left iliac system than the right, which can be distinguished by physical and laboratory examinations. Immediate diagnosis and early management can give an excellent outcome.
The authors thank gynecologists, general and vascular surgeons at the First Affiliated Hospital of Dalian Medical University, Dalian, China, for the supportive information. They also thank Emeritus professor Leon Speroff at Department of Obstetrics and Gynecology, Oregon Health and Science University, Portland, USA, for the language correction.
REFERENCES 1. Tannous H, Nasrallah F, Marjani M. Spontaneous iliac vein rupture: case report and comprehensive review of the literature. Ann Vasc Surg 2006;20:258-262.
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2. Borkon MJ, Dassinger MS, Dattilo JB. Spontaneous iliac vein rupture. Am Surg 2009;75:1151-1152. 3. Hossne WS, Nahas PS, Vasconcelos E. Spontaneous rupture of the iliac vein; acute abdomen. Arq Cir Clin Exp 1961;24: 27-30. 4. Elliot D, Ware CC. Spontaneous rupture of external iliac vein. J R Soc Med 1982;75:477-478.
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5. Steinberg JB, Jacocks MA. May-Thurner syndrome: a previously unreported variant. Ann Vasc Surg 1993;7:577-581. 6. McDonald RT, Vorpahl TE, Caskey J. Spontaneous rupture of the iliac vein. Vasc Surg 1980;14:330-333. 7. Gaschignard N, Le Paul Y, Maouni T, et al. Spontaneous rupture of the left common iliac vein. Ann Vasc Surg 2000;14:517-518.