Behcet disease, Embolization, Pseudoaneurysm, Anterior Tibial Artery

A 52-year-old man

The patient had recurrent oral and genital ulcer and recurrent arthritis and clinical diagnosis of Behcet disease was made. He had undergone resection of right posterior tibial artery pseudoaneurysm 2 years before admission and had a history of coumadin treatment for the deep vein thrombosis in the right lower leg for 3months. PT was 29%, APTT was 85.7sec and CRP was 1.49mg/dL.

A pseudoaneurysm at anterior tibial artery in Behcet disease

Ultrasonography reveals a large pseudoaneurysm with thrombus in it (Fig. 1). Selective left femoral angiogram shows a pseudoaneurysm of anterior tibial artery (Fig. 2A, B). Locating a 5F Cobra catheter(Cook, Bloomingtom, USA) at the proximal portion of the pseudoaneurysm, coils were deployed (Fig. 3A). After embolization, left femoral angiogram reveals retrograde filling of the pseudoaneurysm (arrow) through the distal anterior tibial artery (Fig. 3B). With direct puncture of the distal portion of the anterior tibial artery under the US guidance (Fig. 4A), we could obliterate the distal portion of the pseudoaneurysm with coils (Fig. 4B). Left femoral angiogram after coil embolization shows complete occlusion of the pseudoaneurysm (Fig. 5).

Fig. 1. Ultrasonography reveals a large peudoaneurysm with partial thrombosis.

Fig. 2. Selective left femoral angiogram shows pseudoaneurysm of anteror tibial artery.

Fig. 3. After embolization at the proximal portion of the pseudoaneurysm, left femoral angiogram reveals retrograde filling of the pseudoaneurysm(arrow) through the distal anterior tibial artery(arrowhead).

Fig. 4. With direct puncture of the distal portion of the anterior tibial artery, coils were deployed at distal portion of the pseudoaneurysm.

Fig. 5. Left femoral angiogrm after coil embolization shows complete occlusion of the pseudoaneurysm.

Vascular involvement in Behcet disease may cause life-threatening conditions and the incidence of cardiovascular involvement has been reported as 7%-29%. In this systemic disease, any artery or vein in the body may be affected and the involvement of the vascular tree manifests itself pathologically as arterial occlusion, arterial aneurysm, venous occlusion, and varices. Venous lesions are thought to be more common than arterial ones, but the leading cause of death in patients with Behcet disease is rupture of large aneurysms. Behcet’s aneurysms could involve the whole arterial tree. The most common site of aneurysm formation is the abdominal aorta, followed by the pulmonary, femoral, subclavian, popliteal, common carotid, coronary, brachial, ulnar, common iliac, external iliac, tibial, renal, cerebral, axillary, and splenic arteries. The pathogenesis of aneurysms in Behcet disease is quite different from that of atherosclerotic aneurysms. Inflammatory cells composed of neutrophil, lymphocytes, and plasma cells infiltrate the media and adventitia, mainly around the proliferated vas vasorum. With the destruction of elastic and muscle cells in the media the vessel wall weakens and a process of aneurysmal dilatation starts. Occlusion of the vasa vasorum speeds this process by transmural necrosis. Ultimately, perforation of the vessel wall and pseudoaneurysm formation ensues. Surgical treatment of Behcet aneurysms is often unsuccessful and results in graft occlusions, anastomotic site pseudoaneurysms, and/or new aneurysms. In this case, we decided to treat the pseudoaneurysm with coil embolization. But, after coil embolization at the proximal portion of the pseudoaneurysm due to technical difficulty to traverse the pseudoaneurysm neck, left femoral angiogram reveals retrograde filling of the pseudoaneurysms through the distal anterior tibial artery. With direct puncture of the distal portion of the anterior tibial artery, we could obliterate the distal portion of the pseudoaneurysm successfully with coils.

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