- Email: [email protected]
Nutcracker syndrome and deep venous thrombosis in a patient with duplicated inferior vena cava
Nutcracker syndrome and deep venous thrombosis in a patient with duplicated inferior vena cava Ricardo de Alvarenga Yoshida, MD, PhD,a Winston Bonetti Yoshida, MD, PhD,a Renato Fanchiotti Costa, MD,a Marcelo Souto Nacif, MD, PhD,b Marcone Lima Sobreira, MD, PhD,a and Rodrigo Gibin Jaldin, MD,a São Paulo and Niterói, Brazil Duplicated inferior vena cava is a rare anomaly, and thrombosis in one or both segments is even less frequent. We present a case of deep venous thrombosis of the left lower limb involving the popliteal, femoral, and iliac veins as well as the left segment of the duplicated vena cava and nutcracker syndrome. After catheter-directed thrombolysis
complemented by mechanical thrombolysis, the compromised veins had complete revascularization; the nutcracker syndrome was treated with stent placement, followed by the use of anticoagulants. There was technical success and complete recovery of the patient. (J Vasc Surg: Venous and Lym Dis 2015;-:1-5.)
The most frequent anatomic variations of the vena cava are duplicated inferior vena cava (IVC), retroureteral IVC, left IVC, retroaortic left renal vein or retroaortic IVC, circumaortic renal vein, and IVC draining into azygos vein.1-3 Duplication of the IVC is a rare malformation, with a prevalence of 0.2% to 3%.2,4 Its association with deep venous thrombosis (DVT) or thrombosis of the IVC itself is even rarer.5 Duplication occurs when any of the three paired primitive veins fail to interconnect or regress. The most widely accepted explanation is failure of the subcardinal veins to fuse or failure of the left subcardinal vein to regress.2,6 We present a case of extensive DVT of the left lower extremity and the left limb of the duplicated vena cava associated with extrinsic compression of the left renal vein by the superior mesenteric artery (SMA). The patient was successfully treated with lytic therapy and stenting of the extrinsic compression, followed by the use of anticoagulants drugs, with technical success and complete recovery. We did not find any similar case in the literature.
confirmed extensive venous thrombosis involving the iliac, femoral, and popliteal veins of the left lower limb. Anticoagulant therapy with enoxaparin (60 mg subcutaneously, twice daily) was started. The patient was taking oral contraceptives and denied previous personal or family history of venous thromboembolism. Genetic thrombophilia examinations did not show any abnormalities. After 48 hours with no improvement, venous magnetic resonance angiography was conducted, confirming the venous thrombosis in the popliteal, femoral, and iliac veins; it also demonstrated the presence of duplicated IVC (Fig 1, A, 1-5), with extension of the thrombosis to the left segment of the vena cava until the junction with the left renal vein (Fig 1, A, 2 and 3). In addition, the left renal vein showed signs of extrinsic compression by the SMA (Fig 1, A, 1) and an angle between the aorta and the SMA <45 degrees (Fig 1, B), suggesting the nutcracker syndrome (Fig 1, C, 1). She was then treated with fibrinolytic agents by means of ultrasound-guided percutaneous implantation of multiperforated catheters placed under fluoroscopy in the left popliteal and femoral veins through a popliteal approach and in the iliac vein and left segment of the vena cava through a femoral approach. Preoperative venography (Fig 2, A-C) confirmed extensive thrombosis and showed important collateral circulation in the pelvis and thigh (Fig 2, E and F). In each multiperforated catheter, recombinant tissue plasminogen activator (Actylise, 1 mg/h) was infused during 24 hours. Completion venography (Fig 2, D-F) demonstrated complete lysis of the thrombus in the popliteal vein, subtotal lysis in the femoral vein, and partial lysis in the iliac vein and left segment of the IVC (Fig 2, E and F). Fibrinolysis was then complemented by mechanical lysis of the thrombi by mechanical thrombolysis (pulse-spray technique), using AngioJet (Boston Scientific, Natick, Mass) and infusing heparinized saline under pressure (5000 IU of unfractionated heparin/500 mL). At the end of this treatment, which evolved eventless, follow-up venography (Fig 2, G-I) demonstrated complete recanalization of all the veins treated and absence of collateral circulation (Fig 2, H and I); a significant narrowing of the lumen of the left renal vein was evident, caused by extrinsic compression by the SMA (Fig 2, D),
CASE REPORT A female patient, 24 years old, was referred with important spontaneous asymmetric edema in the left lower limb, 1 day before admission, suggestive of DVT. Color Doppler ultrasound From the Department of Surgery and Orthopedics of the School of Medicine of Botucatu, Universidade Estadual Paulista (UNESP), São Pauloa; and the Radiology Department, Universidade Federal Fluminense (UFF), Niterói.b Author conflict of interest: none. Correspondence: Ricardo de Alvarenga Yoshida, MD, PhD, Depto de Cirurgia e Ortopedia, Faculdade de Medicina de Botucatu, Universidade Estadual Paulista, Campus de Botucatu, Botucatu, São Paulo, Brazil 18618-970 (e-mail: [email protected]). The editors and reviewers of this article have no relevant financial relationships to disclose per the Journal policy that requires reviewers to decline review of any manuscript for which they may have a conflict of interest. 2213-333X Copyright Ó 2015 by the Society for Vascular Surgery. Published by Elsevier Inc. http://dx.doi.org/10.1016/j.jvsv.2015.10.007
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2 Yoshida et al
JOURNAL OF VASCULAR SURGERY: VENOUS AND LYMPHATIC DISORDERS --- 2015
Fig 1. A, 1, Duplicated inferior vena cava (IVC). The left renal vein trajectory is subjacent to the superior mesenteric artery (SMA) with extrinsic compressiondnutcracker syndrome. 2 and 3, Deep venous thrombosis (DVT) to the left segment of the vena cava progressing until the junction with the left renal vein. 4 and 5, Right segment of the vena cava. B, Angle between the aorta and the SMA <45 degrees. C, 1, Left renal vein with extrinsic compression by the SMAdnutcracker syndrome. as previously suggested by magnetic resonance angiography. This stenosis was successfully treated by deployment of a selfexpandable stent (Zilver Vena venous stent 16 40; Cook Medical, Bloomington, Ind) distally to the left renal junction (Fig 2, G). The patient continued anticoagulant therapy (enoxaparin 60 mg subcutaneously, twice daily) for another 2 days, when there was complete remission of the edema and symptoms. She was discharged with oral anticoagulant therapy with rivaroxaban 20 mg/d for 1 year and elastic stockings, with quarterly visits. In the 3-month follow-up, she remains asymptomatic, with no edema in the left lower limb. In the follow-up, both computed tomography angiography and duplex ultrasound showed that all the treated veins were patent, there was no in-stent stenosis (Fig 3, A), and the left renal vein remained patent (Fig 3, B).
DISCUSSION Among the five types of anatomic anomalies of the vena cava,2 type 2A would be the closest to the one presented here. In this type, the two venae cavae originate from the respective iliac veins and progress with no communication between them until the confluence at the point of drainage from the renal veins.2 The smaller angle of the SMA in relation to the aorta caused extrinsic compression of the left renal vein, just like it happens in the classic nutcracker syndrome. We could not find any case previously described with this peculiar anatomy and consequence. The nutcracker syndrome is characterized by the extrinsic compression of the left renal vein by the SMA,
causing venous hypertension, pelvic and renal congestion, and intrarenal and extrarenal or pelvic collateral circulation.7 In our case, the SMA compression was over the left renal vein distally at the confluence with left limb of the IVC. We believe that this was an important additional factor for DVT development and its extension to the deep veins of the left lower limb. Nevertheless, it is difficult to establish with certainty whether in this case the DVT of the iliac-femoral-popliteal sector was a cause or consequence of the thrombosis of the left vena cava; as other risk factors were present, like oral contraceptives, the cause was probably an association of risk factors. Anticoagulation is the treatment of choice for DVT in international guidelines.8 The vena cava filter would be indicated when anticoagulation is contraindicated or bleeding or pulmonary thromboembolism occurs with its use. There are reports of single or double implants,5,9,10 suprarenal or infrarenal,4,11 in duplicated vena cava cases. For young patients with important symptoms and no apparent risk for bleeding, fibrinolytic treatment is the treatment of choice.12,13 In a literature review, with 979 limbs in 11 papers,13 patency rates of thrombolysis in the iliac-femoral veins varied from 60% to 95% after 6 to 12 months and 82% after 6 years, with a mortality rate of <1% as well as early relief of the symptoms and reduction of the risk of chronic venous insufficiency. In our case, this treatment option was successful and uneventful. The venous compression caused by the SMA can be treated by deployment of a self-expandable stent at the site of the compression14 or left renal vein transposition.15
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Yoshida et al 3
Fig 2. A-C, Preoperative venography confirmed extensive thrombosis and substantial collateral circulation in the pelvis and thigh. D, The 24-hour follow-up venogram after thrombolysis: significant narrowing of the lumen of the left renal vein was evident, caused by extrinsic compression by the superior mesenteric artery (SMA). E and F, The 24-hour follow-up venogram after thrombolysis: complete lysis of the thrombus in the popliteal vein, subtotal lysis in the femoral vein, and partial lysis in the iliac vein and left segment of the inferior vena cava (IVC). G, Extrinsic compression treated by deployment of a self-expandable stent (Zilver Vena venous stent 16 40; Cook Medical). H and I, Venogram after mechanical lysis: complete recanalization of all the veins treated and absence of collateral circulation.
4 Yoshida et al
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Fig 3. Computed tomography angiography at 3 months of follow-up. A, Left vena cava and iliac and femoral veins were patent, and there was no in-stent stenosis. B, Left renal vein patent.
Some technical aspects deserve additional consideration. (1) Zilver Vena stent is approved for endovascular treatment of venous diseases in our country and has advantages over the Wallstent (Boston Scientific) because of its higher radial force and less foreshortening, reducing migration risk and increasing delivery precision, respectively. (2) The shortest extension of Zilver Vena stents is 40 mm. To avoid covering the left renal vein, the option was to extend part of the stent into the right vena cava. Encroaching the stent in the vena cava is a common practice in May-Thurner syndrome endovascular treatment. Jarrett et al16 did not find an impact on blood flow with this technique in their series. Our patient also did not show any problem in the followup. (3) Intravascular ultrasound certainly would improve precision in stent delivery and stenosis confirmation, but it was not available in our service. (4) Despite the good long-term results of open surgery, it has been associated with elevated risk of reinterventions (27%) and venous thrombosis.15 Besides, the patient had already been treated by an endovascular procedure for thrombolysis, so the subsequent endovascular approach for treatment of nutcracker syndrome was preferred in this case. In a recent metaanalysis, venous stenting was considered relatively effective and safe for post-thrombotic syndrome and nonthrombotic iliac vein lesions, with low incidence of complications.17 Stenting to treat nutcracker syndrome was performed to avoid recurrence of DVT, and oral anticoagulant therapy was prescribed in the follow-up to ensure the patency of the venous recanalization, although there is no evidence in the literature favoring antiplatelets or anticoagulants
after venous stenting. Our preference was for anticoagulation and its extension for 12 months. CONCLUSIONS Duplicated vena cava DVT with compression by the SMA simulating nutcracker syndrome, with extension of the thrombosis into deep veins of the thigh, is an unusual situation. Fibrinolytic treatment complemented by mechanical lysis (pulse-spray), associated with implantation of a stent at the site of the extrinsic compression, was an efficient and safe treatment in this case. REFERENCES 1. Nanda S, Bhatt SP, Turki MA. Inferior vena cava anomaliesda common cause of DVT and PE commonly not diagnosed. Am J Med Sci 2008;335:409-10. 2. Chen H, Emura S, Nagasaki S, Kubo KY. Double inferior vena cava with interiliac vein: a case report and literature review. Okajimas Folia Anat Jpn 2012;88:147-51. 3. Qian ZY, Yang MF, Zuo KQ, Cheng J, Xiao HB, Ding WX. Computed tomography manifestations of common inferior vena cava dysplasia and its clinical significance. Exp Ther Med 2013;5:631-5. 4. Mano A, Tatsumi T, Sakai H, Imoto Y, Nomura T, Nishikawa S, et al. A case of deep venous thrombosis with a double inferior vena cava effectively treated by suprarenal filter implantation. Jpn Heart J 2004;45:1063-9. 5. Sartori MT, Zampieri P, Andres AL, Prandoni P, Motta R, Miotto D. Double vena cava filter insertion in congenital duplicated inferior vena cava: a case report and literature review. Haematologica 2006;91(Suppl): ECR30. 6. Babu CS, Lalwani R, Kumar I. Right double inferior vena cava (IVC) with preaortic iliac confluencedcase report and review of literature. J Clin Diagn Res 2014;8:130-2.
JOURNAL OF VASCULAR SURGERY: VENOUS AND LYMPHATIC DISORDERS Volume -, Number -
7. Rudloff U, Holmes RJ, Prem JT, Faust GR, Moldwin R, Siegel D. Mesoaortic compression of the left renal vein (nutcracker syndrome): case reports and review of the literature. Ann Vasc Surg 2006;20:120-9. 8. Kearon C, Akl EA, Comerota AJ, Prandoni P, Bounameaux H, Goldhaber SZ, et al. Antithrombotic therapy for VTE disease: Antithrombotic Therapy and Prevention of Thrombosis, 9th ed: American College of Chest Physicians Evidence-Based Clinical Practice Guidelines. Chest 2012;141(Suppl):e419S-94S. 9. Pena-Duque MA, Damas-de-los-Santos F, Villavicencio-Fernandez R, Martinez-Rios MA. [Thrombosis of double inferior vena cava with a thrombosis-in-transit and insertion of two filters]. Rev Invest Clin 2010;62:279-80. 10. Siddiqui RA, Hans S. Double inferior vena cava filter implantation in a patient with a duplicate inferior vena cava. J Invasive Cardiol 2008;20: 91-2. 11. Malgor RD, Sobreira ML, Boaventura PN, Moura R, Yoshida WB. Filter placement in duplicated inferior vena cava: case report and review of the literature. J Vasc Bras 2008;7:167-70. 12. Wang X, Chen Z, Cai Q. Catheter-directed thrombolysis for double inferior vena cava with deep venous thrombosis: a case report and literature review. Phlebology 2014;29:480-3.
Yoshida et al 5
13. Broholm R, Panduro Jensen L, Baekgaard N. Catheter-directed thrombolysis in the treatment of iliofemoral venous thrombosis. A review. Int Angiol 2010;29:292-302. 14. da Cunha Júnior JR, de Souza TC, Feitosa AT, Brizzi JR, Tinoco JA. Endovascular treatment of nutcracker syndrome. J Vasc Bras 2013;12: 247-51. 15. Reed NR, Kalra M, Bower TC, Vrtiska TJ, Ricotta JJ 2nd, Gloviczki P. Left renal vein transposition for nutcracker syndrome. J Vasc Surg 2009;49:386-93; discussion: 393-4. 16. Jarrett H, Zack CJ, Aggarwal V, Lakhter V, Alkhouli MA, Zhao H, et al. Impact of institutional volume on outcomes of catheter directed thrombolysis in the treatment of acute proximal deep vein thrombosis: a 6-year United States experience (2005-2010). Circulation 2015;132: 1127-35. 17. Wen-da W, Yu Z, Yue-Xin C. Stenting for chronic obstructive venous disease: a current comprehensive meta-analysis and systematic review. Phlebology 2015; [Epub ahead of print].
Submitted Jul 31, 2015; accepted Oct 11, 2015.
complemented by mechanical thrombolysis, the compromised veins had complete revascularization; the nutcracker syndrome was treated with stent placement, followed by the use of anticoagulants. There was technical success and complete recovery of the patient. (J Vasc Surg: Venous and Lym Dis 2015;-:1-5.)
The most frequent anatomic variations of the vena cava are duplicated inferior vena cava (IVC), retroureteral IVC, left IVC, retroaortic left renal vein or retroaortic IVC, circumaortic renal vein, and IVC draining into azygos vein.1-3 Duplication of the IVC is a rare malformation, with a prevalence of 0.2% to 3%.2,4 Its association with deep venous thrombosis (DVT) or thrombosis of the IVC itself is even rarer.5 Duplication occurs when any of the three paired primitive veins fail to interconnect or regress. The most widely accepted explanation is failure of the subcardinal veins to fuse or failure of the left subcardinal vein to regress.2,6 We present a case of extensive DVT of the left lower extremity and the left limb of the duplicated vena cava associated with extrinsic compression of the left renal vein by the superior mesenteric artery (SMA). The patient was successfully treated with lytic therapy and stenting of the extrinsic compression, followed by the use of anticoagulants drugs, with technical success and complete recovery. We did not find any similar case in the literature.
confirmed extensive venous thrombosis involving the iliac, femoral, and popliteal veins of the left lower limb. Anticoagulant therapy with enoxaparin (60 mg subcutaneously, twice daily) was started. The patient was taking oral contraceptives and denied previous personal or family history of venous thromboembolism. Genetic thrombophilia examinations did not show any abnormalities. After 48 hours with no improvement, venous magnetic resonance angiography was conducted, confirming the venous thrombosis in the popliteal, femoral, and iliac veins; it also demonstrated the presence of duplicated IVC (Fig 1, A, 1-5), with extension of the thrombosis to the left segment of the vena cava until the junction with the left renal vein (Fig 1, A, 2 and 3). In addition, the left renal vein showed signs of extrinsic compression by the SMA (Fig 1, A, 1) and an angle between the aorta and the SMA <45 degrees (Fig 1, B), suggesting the nutcracker syndrome (Fig 1, C, 1). She was then treated with fibrinolytic agents by means of ultrasound-guided percutaneous implantation of multiperforated catheters placed under fluoroscopy in the left popliteal and femoral veins through a popliteal approach and in the iliac vein and left segment of the vena cava through a femoral approach. Preoperative venography (Fig 2, A-C) confirmed extensive thrombosis and showed important collateral circulation in the pelvis and thigh (Fig 2, E and F). In each multiperforated catheter, recombinant tissue plasminogen activator (Actylise, 1 mg/h) was infused during 24 hours. Completion venography (Fig 2, D-F) demonstrated complete lysis of the thrombus in the popliteal vein, subtotal lysis in the femoral vein, and partial lysis in the iliac vein and left segment of the IVC (Fig 2, E and F). Fibrinolysis was then complemented by mechanical lysis of the thrombi by mechanical thrombolysis (pulse-spray technique), using AngioJet (Boston Scientific, Natick, Mass) and infusing heparinized saline under pressure (5000 IU of unfractionated heparin/500 mL). At the end of this treatment, which evolved eventless, follow-up venography (Fig 2, G-I) demonstrated complete recanalization of all the veins treated and absence of collateral circulation (Fig 2, H and I); a significant narrowing of the lumen of the left renal vein was evident, caused by extrinsic compression by the SMA (Fig 2, D),
CASE REPORT A female patient, 24 years old, was referred with important spontaneous asymmetric edema in the left lower limb, 1 day before admission, suggestive of DVT. Color Doppler ultrasound From the Department of Surgery and Orthopedics of the School of Medicine of Botucatu, Universidade Estadual Paulista (UNESP), São Pauloa; and the Radiology Department, Universidade Federal Fluminense (UFF), Niterói.b Author conflict of interest: none. Correspondence: Ricardo de Alvarenga Yoshida, MD, PhD, Depto de Cirurgia e Ortopedia, Faculdade de Medicina de Botucatu, Universidade Estadual Paulista, Campus de Botucatu, Botucatu, São Paulo, Brazil 18618-970 (e-mail: [email protected]). The editors and reviewers of this article have no relevant financial relationships to disclose per the Journal policy that requires reviewers to decline review of any manuscript for which they may have a conflict of interest. 2213-333X Copyright Ó 2015 by the Society for Vascular Surgery. Published by Elsevier Inc. http://dx.doi.org/10.1016/j.jvsv.2015.10.007
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2 Yoshida et al
JOURNAL OF VASCULAR SURGERY: VENOUS AND LYMPHATIC DISORDERS --- 2015
Fig 1. A, 1, Duplicated inferior vena cava (IVC). The left renal vein trajectory is subjacent to the superior mesenteric artery (SMA) with extrinsic compressiondnutcracker syndrome. 2 and 3, Deep venous thrombosis (DVT) to the left segment of the vena cava progressing until the junction with the left renal vein. 4 and 5, Right segment of the vena cava. B, Angle between the aorta and the SMA <45 degrees. C, 1, Left renal vein with extrinsic compression by the SMAdnutcracker syndrome. as previously suggested by magnetic resonance angiography. This stenosis was successfully treated by deployment of a selfexpandable stent (Zilver Vena venous stent 16 40; Cook Medical, Bloomington, Ind) distally to the left renal junction (Fig 2, G). The patient continued anticoagulant therapy (enoxaparin 60 mg subcutaneously, twice daily) for another 2 days, when there was complete remission of the edema and symptoms. She was discharged with oral anticoagulant therapy with rivaroxaban 20 mg/d for 1 year and elastic stockings, with quarterly visits. In the 3-month follow-up, she remains asymptomatic, with no edema in the left lower limb. In the follow-up, both computed tomography angiography and duplex ultrasound showed that all the treated veins were patent, there was no in-stent stenosis (Fig 3, A), and the left renal vein remained patent (Fig 3, B).
DISCUSSION Among the five types of anatomic anomalies of the vena cava,2 type 2A would be the closest to the one presented here. In this type, the two venae cavae originate from the respective iliac veins and progress with no communication between them until the confluence at the point of drainage from the renal veins.2 The smaller angle of the SMA in relation to the aorta caused extrinsic compression of the left renal vein, just like it happens in the classic nutcracker syndrome. We could not find any case previously described with this peculiar anatomy and consequence. The nutcracker syndrome is characterized by the extrinsic compression of the left renal vein by the SMA,
causing venous hypertension, pelvic and renal congestion, and intrarenal and extrarenal or pelvic collateral circulation.7 In our case, the SMA compression was over the left renal vein distally at the confluence with left limb of the IVC. We believe that this was an important additional factor for DVT development and its extension to the deep veins of the left lower limb. Nevertheless, it is difficult to establish with certainty whether in this case the DVT of the iliac-femoral-popliteal sector was a cause or consequence of the thrombosis of the left vena cava; as other risk factors were present, like oral contraceptives, the cause was probably an association of risk factors. Anticoagulation is the treatment of choice for DVT in international guidelines.8 The vena cava filter would be indicated when anticoagulation is contraindicated or bleeding or pulmonary thromboembolism occurs with its use. There are reports of single or double implants,5,9,10 suprarenal or infrarenal,4,11 in duplicated vena cava cases. For young patients with important symptoms and no apparent risk for bleeding, fibrinolytic treatment is the treatment of choice.12,13 In a literature review, with 979 limbs in 11 papers,13 patency rates of thrombolysis in the iliac-femoral veins varied from 60% to 95% after 6 to 12 months and 82% after 6 years, with a mortality rate of <1% as well as early relief of the symptoms and reduction of the risk of chronic venous insufficiency. In our case, this treatment option was successful and uneventful. The venous compression caused by the SMA can be treated by deployment of a self-expandable stent at the site of the compression14 or left renal vein transposition.15
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Yoshida et al 3
Fig 2. A-C, Preoperative venography confirmed extensive thrombosis and substantial collateral circulation in the pelvis and thigh. D, The 24-hour follow-up venogram after thrombolysis: significant narrowing of the lumen of the left renal vein was evident, caused by extrinsic compression by the superior mesenteric artery (SMA). E and F, The 24-hour follow-up venogram after thrombolysis: complete lysis of the thrombus in the popliteal vein, subtotal lysis in the femoral vein, and partial lysis in the iliac vein and left segment of the inferior vena cava (IVC). G, Extrinsic compression treated by deployment of a self-expandable stent (Zilver Vena venous stent 16 40; Cook Medical). H and I, Venogram after mechanical lysis: complete recanalization of all the veins treated and absence of collateral circulation.
4 Yoshida et al
JOURNAL OF VASCULAR SURGERY: VENOUS AND LYMPHATIC DISORDERS --- 2015
Fig 3. Computed tomography angiography at 3 months of follow-up. A, Left vena cava and iliac and femoral veins were patent, and there was no in-stent stenosis. B, Left renal vein patent.
Some technical aspects deserve additional consideration. (1) Zilver Vena stent is approved for endovascular treatment of venous diseases in our country and has advantages over the Wallstent (Boston Scientific) because of its higher radial force and less foreshortening, reducing migration risk and increasing delivery precision, respectively. (2) The shortest extension of Zilver Vena stents is 40 mm. To avoid covering the left renal vein, the option was to extend part of the stent into the right vena cava. Encroaching the stent in the vena cava is a common practice in May-Thurner syndrome endovascular treatment. Jarrett et al16 did not find an impact on blood flow with this technique in their series. Our patient also did not show any problem in the followup. (3) Intravascular ultrasound certainly would improve precision in stent delivery and stenosis confirmation, but it was not available in our service. (4) Despite the good long-term results of open surgery, it has been associated with elevated risk of reinterventions (27%) and venous thrombosis.15 Besides, the patient had already been treated by an endovascular procedure for thrombolysis, so the subsequent endovascular approach for treatment of nutcracker syndrome was preferred in this case. In a recent metaanalysis, venous stenting was considered relatively effective and safe for post-thrombotic syndrome and nonthrombotic iliac vein lesions, with low incidence of complications.17 Stenting to treat nutcracker syndrome was performed to avoid recurrence of DVT, and oral anticoagulant therapy was prescribed in the follow-up to ensure the patency of the venous recanalization, although there is no evidence in the literature favoring antiplatelets or anticoagulants
after venous stenting. Our preference was for anticoagulation and its extension for 12 months. CONCLUSIONS Duplicated vena cava DVT with compression by the SMA simulating nutcracker syndrome, with extension of the thrombosis into deep veins of the thigh, is an unusual situation. Fibrinolytic treatment complemented by mechanical lysis (pulse-spray), associated with implantation of a stent at the site of the extrinsic compression, was an efficient and safe treatment in this case. REFERENCES 1. Nanda S, Bhatt SP, Turki MA. Inferior vena cava anomaliesda common cause of DVT and PE commonly not diagnosed. Am J Med Sci 2008;335:409-10. 2. Chen H, Emura S, Nagasaki S, Kubo KY. Double inferior vena cava with interiliac vein: a case report and literature review. Okajimas Folia Anat Jpn 2012;88:147-51. 3. Qian ZY, Yang MF, Zuo KQ, Cheng J, Xiao HB, Ding WX. Computed tomography manifestations of common inferior vena cava dysplasia and its clinical significance. Exp Ther Med 2013;5:631-5. 4. Mano A, Tatsumi T, Sakai H, Imoto Y, Nomura T, Nishikawa S, et al. A case of deep venous thrombosis with a double inferior vena cava effectively treated by suprarenal filter implantation. Jpn Heart J 2004;45:1063-9. 5. Sartori MT, Zampieri P, Andres AL, Prandoni P, Motta R, Miotto D. Double vena cava filter insertion in congenital duplicated inferior vena cava: a case report and literature review. Haematologica 2006;91(Suppl): ECR30. 6. Babu CS, Lalwani R, Kumar I. Right double inferior vena cava (IVC) with preaortic iliac confluencedcase report and review of literature. J Clin Diagn Res 2014;8:130-2.
JOURNAL OF VASCULAR SURGERY: VENOUS AND LYMPHATIC DISORDERS Volume -, Number -
7. Rudloff U, Holmes RJ, Prem JT, Faust GR, Moldwin R, Siegel D. Mesoaortic compression of the left renal vein (nutcracker syndrome): case reports and review of the literature. Ann Vasc Surg 2006;20:120-9. 8. Kearon C, Akl EA, Comerota AJ, Prandoni P, Bounameaux H, Goldhaber SZ, et al. Antithrombotic therapy for VTE disease: Antithrombotic Therapy and Prevention of Thrombosis, 9th ed: American College of Chest Physicians Evidence-Based Clinical Practice Guidelines. Chest 2012;141(Suppl):e419S-94S. 9. Pena-Duque MA, Damas-de-los-Santos F, Villavicencio-Fernandez R, Martinez-Rios MA. [Thrombosis of double inferior vena cava with a thrombosis-in-transit and insertion of two filters]. Rev Invest Clin 2010;62:279-80. 10. Siddiqui RA, Hans S. Double inferior vena cava filter implantation in a patient with a duplicate inferior vena cava. J Invasive Cardiol 2008;20: 91-2. 11. Malgor RD, Sobreira ML, Boaventura PN, Moura R, Yoshida WB. Filter placement in duplicated inferior vena cava: case report and review of the literature. J Vasc Bras 2008;7:167-70. 12. Wang X, Chen Z, Cai Q. Catheter-directed thrombolysis for double inferior vena cava with deep venous thrombosis: a case report and literature review. Phlebology 2014;29:480-3.
Yoshida et al 5
13. Broholm R, Panduro Jensen L, Baekgaard N. Catheter-directed thrombolysis in the treatment of iliofemoral venous thrombosis. A review. Int Angiol 2010;29:292-302. 14. da Cunha Júnior JR, de Souza TC, Feitosa AT, Brizzi JR, Tinoco JA. Endovascular treatment of nutcracker syndrome. J Vasc Bras 2013;12: 247-51. 15. Reed NR, Kalra M, Bower TC, Vrtiska TJ, Ricotta JJ 2nd, Gloviczki P. Left renal vein transposition for nutcracker syndrome. J Vasc Surg 2009;49:386-93; discussion: 393-4. 16. Jarrett H, Zack CJ, Aggarwal V, Lakhter V, Alkhouli MA, Zhao H, et al. Impact of institutional volume on outcomes of catheter directed thrombolysis in the treatment of acute proximal deep vein thrombosis: a 6-year United States experience (2005-2010). Circulation 2015;132: 1127-35. 17. Wen-da W, Yu Z, Yue-Xin C. Stenting for chronic obstructive venous disease: a current comprehensive meta-analysis and systematic review. Phlebology 2015; [Epub ahead of print].
Submitted Jul 31, 2015; accepted Oct 11, 2015.