Ann Phlebology 2022; 20(2): 104-107
Published online December 31, 2022
https://doi.org/10.37923/phle.2022.20.2.104
© Annals of phlebology
Correspondence to : Tae-geun Rim, 1262, 2sunhwan-ro, Heungdeokgu, Cheongju 28378, Korea, Department of Thoraic and Cardiovascular Surgery, Cheongju HANA Hostpital
Tel: 043-230-6559, Fax: 043-235-5300
E-mail: oranutan@naver.com
An 82-year-old female was referred from a local clinic due to left leg swelling despite 1 month of direct oral anticoagulant therapy for left iliac deep vein thrombosis. Enhanced computed tomography (CT) revealed a hypoplastic iliac vein with several iliac and popliteal arteriovenous fistulas (AVFs), without significant iliofemoral deep vein thrombosis. First, endovascular balloon angioplasty was performed for iliac vein hypoplasia related to a previous deep vein thrombosis. On postoperative day 4, CT angiography was performed due to persistent left leg edema with circumference discrepancy, which confirmed narrowing of the left common iliac vein with extrinsic compression between the right common iliac artery and vertebral body. Persistent left leg edema required an additional endovascular procedure; more specifically, placement of an iliac vein stent. After restoration of circumference equality in both the thigh and calf, the patient was discharged from hospital on antiplatelet and antithrombotic medications. Three months later, the patient visited again due to recurrent left leg edema, with a circumference discrepancy measuring >10 cm. Iliac vein balloon angioplasty was performed, as was additional stent placement for May-Thurner syndrome (MTS) and coil embolization of the iliac AVF. Endovascular embolization resulted in full recovery from MTS with coexisting iliac AVFs. MTS combined with ilio-iliac AVF(s) may be a neglected phenomenon, although it is not very rare. The present report reviews this disease category and raises awareness of possible disease mechanisms(s).
Keywords May-Thurner’s syndrome, Iliac AVF, Endovascular, Co-existing fistula
May-Thurner syndrome (MTS) does not commonly cause leg edema, pain, or deep vein thrombosis (DVT). However, general understanding of the cause of MTS is left common iliac vein compression by the right common iliac artery and lumbar vertebral body (
An 82-year-old female was referred from a local clinic due to persistent left leg edema caused by subacute onset of DVT. She experienced leg edema for 1 year and recent aggravation for 1 month. The patient had diabetes mellitus, hypertension, and dementia, with no history of spinal or gynecological surgery. The patient’s vital signs were unremarkable, although she exhibited a leg circumference discrepancy of 4 cm, with pretibial pitting edema.
The patient underwent ultrasound examination at a local clinic, which revealed DVT in the left iliac vein. She was administered a direct oral anticoagulant (rivaroxaban, 20 mg sod) and prescribed medical compression stockings for 1 month. Computed tomography (CT) angiography revealed DVT in the left popliteal vein, with a totally collapsed iliac vein and leg swelling. Early filling sign of the left femoral vein was also noted.
Femoral venography revealed total occlusion of the left iliofemoral venous system (Fig. 1A). The patient was treated using percutaneous transluminal angioplasty (PTA) alone (Fig. 1B-D). After the first procedure, the patient’s leg circumference discrepancy was not immediately corrected. Follow-up CT angiography revealed significant extrinsic compression of the left common iliac vein, which is a characteristic finding of MTS. An additional procedure involved stent placement in the common iliac vein using a 10×80 mm self-expandable stent (Boston Scientific, Marlborough, MA, USA) (Fig. 2). This additional procedure resulted in full resolution of left leg edema without a circumference discrepancy. The patient was administered a direct oral anticoagulant and prescribed medical compres-sion stockings after the procedure. Three months after the procedure, however, the patient visited the clinic again due to recurrent left leg edema. The authors decided on a combined procedure address to MTS and AVF. The MTS management maneuver included PTA and placement of an additional stent for in-stent occlusion and AVF closure using coil embolization (Fig. 3). Arteriography of the left lower extremity was performed via right common femoral artery puncture. Selective catheterization of the left internal iliac artery revealed an arteriovenous connection from one branch that was successfully coiled using a 3∼4 mm coil (Boston Scientific). Completion angiography demonstrated significantly improved residual—but minimally delayed—filling. Subsequently, the patient’s leg edema resolved. Comparison of leg circumference was remarkable (at admission, right thigh 37.2 cm; left thigh, 38.2 cm; right calf, 27.6 cm; left calf, 33.4 cm; at discharge, 42, 41, 26.5, and 26.5 cm, respectively).
MTS is a condition in which the left common iliac vein is compressed by the right common iliac artery and the lumbar vertebra. This may have caused swelling or DVT in the left lower extremity. MTS was first described by May and Thurner in 1957 (
The etiology of ilio-iliac AVF(s) is variable and includes congenital, iatrogenic, and trauma. However, MTS coexisting with left iliac AVFs is a rare phenomenon. In this case, there was no aneurysm, catheter insertion, or history of trauma. It is very difficult to establish etiological relationships. In rare cases of AVF formation within a thrombosed vein, it has been hypothesized that hypoxia, shear stress, and venous hypertension result in the upregulation of hypoxia-induced microvascular growth (
Conventionally, some authors recommend small-size, temporary femoral AVF formation that is helpful in maintaining the open state of the stent on the iliac vein. Therefore, many vascular surgeons neglect the presence of small-size AVFs. AVFs may have inhibited antegrade venous blood flow due to the Venturi effect and worsened edema in the lower extremities (
Haijie et al. reported that embolization resulted in decreased venous pressure, and opening and stenting of the occlusion of the iliac vein further reduced venous flow resistance (
The patient described in the present report was treated using a general strategy for MTS; more specifically, PTA with stenting first, and addressing coexisting left iliac AVFs. Recurrence of DVT with leg edema forced AVF closure using an endovascular procedure.
Coexisting AVF with MTS may be an overlooked lesion. Clinicians should consider the presence of coexisting vascular anomalies when managing patients with MTS. Endovascular therapy using embolization and stenting can provide adequate resolution of these lesions. To date, the pathogenic mechanism of spontaneous-onset AVFs coexisting with MTS remains unclear. This report may raise awareness of these vascular lesions among vascular surgeons in the field.
Ann Phlebology 2022; 20(2): 104-107
Published online December 31, 2022 https://doi.org/10.37923/phle.2022.20.2.104
Copyright © Annals of phlebology.
Tae-geun Rim, M.D.
Department of Thoraic and Cardiovascular Surgery, Cheongju HANA Hostpital, Cheongju, Korea
Correspondence to:Tae-geun Rim, 1262, 2sunhwan-ro, Heungdeokgu, Cheongju 28378, Korea, Department of Thoraic and Cardiovascular Surgery, Cheongju HANA Hostpital
Tel: 043-230-6559, Fax: 043-235-5300
E-mail: oranutan@naver.com
An 82-year-old female was referred from a local clinic due to left leg swelling despite 1 month of direct oral anticoagulant therapy for left iliac deep vein thrombosis. Enhanced computed tomography (CT) revealed a hypoplastic iliac vein with several iliac and popliteal arteriovenous fistulas (AVFs), without significant iliofemoral deep vein thrombosis. First, endovascular balloon angioplasty was performed for iliac vein hypoplasia related to a previous deep vein thrombosis. On postoperative day 4, CT angiography was performed due to persistent left leg edema with circumference discrepancy, which confirmed narrowing of the left common iliac vein with extrinsic compression between the right common iliac artery and vertebral body. Persistent left leg edema required an additional endovascular procedure; more specifically, placement of an iliac vein stent. After restoration of circumference equality in both the thigh and calf, the patient was discharged from hospital on antiplatelet and antithrombotic medications. Three months later, the patient visited again due to recurrent left leg edema, with a circumference discrepancy measuring >10 cm. Iliac vein balloon angioplasty was performed, as was additional stent placement for May-Thurner syndrome (MTS) and coil embolization of the iliac AVF. Endovascular embolization resulted in full recovery from MTS with coexisting iliac AVFs. MTS combined with ilio-iliac AVF(s) may be a neglected phenomenon, although it is not very rare. The present report reviews this disease category and raises awareness of possible disease mechanisms(s).
Keywords: May-Thurner&rsquo,s syndrome, Iliac AVF, Endovascular, Co-existing fistula
May-Thurner syndrome (MTS) does not commonly cause leg edema, pain, or deep vein thrombosis (DVT). However, general understanding of the cause of MTS is left common iliac vein compression by the right common iliac artery and lumbar vertebral body (
An 82-year-old female was referred from a local clinic due to persistent left leg edema caused by subacute onset of DVT. She experienced leg edema for 1 year and recent aggravation for 1 month. The patient had diabetes mellitus, hypertension, and dementia, with no history of spinal or gynecological surgery. The patient’s vital signs were unremarkable, although she exhibited a leg circumference discrepancy of 4 cm, with pretibial pitting edema.
The patient underwent ultrasound examination at a local clinic, which revealed DVT in the left iliac vein. She was administered a direct oral anticoagulant (rivaroxaban, 20 mg sod) and prescribed medical compression stockings for 1 month. Computed tomography (CT) angiography revealed DVT in the left popliteal vein, with a totally collapsed iliac vein and leg swelling. Early filling sign of the left femoral vein was also noted.
Femoral venography revealed total occlusion of the left iliofemoral venous system (Fig. 1A). The patient was treated using percutaneous transluminal angioplasty (PTA) alone (Fig. 1B-D). After the first procedure, the patient’s leg circumference discrepancy was not immediately corrected. Follow-up CT angiography revealed significant extrinsic compression of the left common iliac vein, which is a characteristic finding of MTS. An additional procedure involved stent placement in the common iliac vein using a 10×80 mm self-expandable stent (Boston Scientific, Marlborough, MA, USA) (Fig. 2). This additional procedure resulted in full resolution of left leg edema without a circumference discrepancy. The patient was administered a direct oral anticoagulant and prescribed medical compres-sion stockings after the procedure. Three months after the procedure, however, the patient visited the clinic again due to recurrent left leg edema. The authors decided on a combined procedure address to MTS and AVF. The MTS management maneuver included PTA and placement of an additional stent for in-stent occlusion and AVF closure using coil embolization (Fig. 3). Arteriography of the left lower extremity was performed via right common femoral artery puncture. Selective catheterization of the left internal iliac artery revealed an arteriovenous connection from one branch that was successfully coiled using a 3∼4 mm coil (Boston Scientific). Completion angiography demonstrated significantly improved residual—but minimally delayed—filling. Subsequently, the patient’s leg edema resolved. Comparison of leg circumference was remarkable (at admission, right thigh 37.2 cm; left thigh, 38.2 cm; right calf, 27.6 cm; left calf, 33.4 cm; at discharge, 42, 41, 26.5, and 26.5 cm, respectively).
MTS is a condition in which the left common iliac vein is compressed by the right common iliac artery and the lumbar vertebra. This may have caused swelling or DVT in the left lower extremity. MTS was first described by May and Thurner in 1957 (
The etiology of ilio-iliac AVF(s) is variable and includes congenital, iatrogenic, and trauma. However, MTS coexisting with left iliac AVFs is a rare phenomenon. In this case, there was no aneurysm, catheter insertion, or history of trauma. It is very difficult to establish etiological relationships. In rare cases of AVF formation within a thrombosed vein, it has been hypothesized that hypoxia, shear stress, and venous hypertension result in the upregulation of hypoxia-induced microvascular growth (
Conventionally, some authors recommend small-size, temporary femoral AVF formation that is helpful in maintaining the open state of the stent on the iliac vein. Therefore, many vascular surgeons neglect the presence of small-size AVFs. AVFs may have inhibited antegrade venous blood flow due to the Venturi effect and worsened edema in the lower extremities (
Haijie et al. reported that embolization resulted in decreased venous pressure, and opening and stenting of the occlusion of the iliac vein further reduced venous flow resistance (
The patient described in the present report was treated using a general strategy for MTS; more specifically, PTA with stenting first, and addressing coexisting left iliac AVFs. Recurrence of DVT with leg edema forced AVF closure using an endovascular procedure.
Coexisting AVF with MTS may be an overlooked lesion. Clinicians should consider the presence of coexisting vascular anomalies when managing patients with MTS. Endovascular therapy using embolization and stenting can provide adequate resolution of these lesions. To date, the pathogenic mechanism of spontaneous-onset AVFs coexisting with MTS remains unclear. This report may raise awareness of these vascular lesions among vascular surgeons in the field.
Insoo Park, M.D., Junseong Kwon, M.D. and Sujin Park, M.D.
Ann Phlebology 2022; 20(2): 111-112