Liver angiography Hepatic arteries, therapeutic blockade Liver, Interventional procedure

33-year-old man

A 33-year-old man with Behcet's disease presented to us with right upper abdominal pain that he had experienced for one day, and so he was admitted to our hospital. There was no previous history of trauma. He was diagnosed with Behcet's disease 3 years ago at a different hospital and diagnosis was confirmed with symptoms of recurrent oral and genital ulcerations, and relapsing uveitis. The patient did not display fever or chills. The initial hemoglobin was 11.3g/dl and the hematocrit was 34.1%.

Behcet's disease

Unenhanced and enhanced abdomen CT was then performed. The CT showed a large subcapsular hematoma of mixed attenuation at the lateral and posterior aspects of the right lobe of the liver (Fig. 1). As a result, the liver was compressed by hematoma. There was a small high attenuating focus within the hematoma compatible with extravasation of the contrast material and active bleeding (Fig. 1B). Angiography and embolization of the aneurysm was then planned. A 5F Yashiro type catheter (Glidecath, Terumo, Tokyo, Japan) was placed via the femoral artery approach at the superior mesenteric artery (SMA). The SMA angiogram showed a small aneurysm in the segment 6 branch of the right hepatic artery arising from the SMA, and there was also extravasation of the contrast material (Fig. 2A). The selective hepatic angiogram that was done using 2.4F microcatheter (Progreat, Terumo, Tokyo, Japan) clearly showed about a 7.8 mm x 3 mm aneurysm.

2.4 F microcatheter was changed with 2F microcatheter (Progreat, Terumo, Tokyo, Japan) for small diameter of the segment 6 branch of the right hepatic artery and placed at the segment 6 branch of the right hepatic artery. Incidentally, one microcoil migrated to the segment 7 branch of the right hepatic artery during embolization, which may be due to inadequate advancement of microcatheter to the segment 6 branch of the right hepatic artery. After further advancement of microcatheter to the segment 6 branch of the right hepatic artery, the aneurysm was successfully embolized by using two microcoils (2mm, Hilal coil, Cook, Bloomington, IN, U.S.A.). The hepatic and SMA angiograms obtained after embolization of the segment 6 branch of the right hepatic artery with microcoils showed that the aneurysm was completely occluded and the extravasation of the contrast materials was no longer seen. An incidentally migrated coil was noted in the segment 7 branch of the right hepatic artery (Fig. 2B). After a week, the abdomen CT showed a decreased amount of subcapsular hematoma in the right liver and there was no evidence of active bleeding. The patient was discharged 1 day later. After 2 months, an abdomen CT was performed again, It showed the greatly decreased size of the subcapsular hematoma of the liver (Fig. 3). One year clinical follow up showed normal findings.

Contrast enhanced abdomen CT (A, B) shows a large hematoma (arrows) of mixed attenuation at the lateral and posterior aspects of the right lobe of the liver along with compression of the adjacent liver. (B) A small high attenuating focus (arrowhead) is noted within the hematoma that suggests extravasation of the contrast material and active bleeding.

A. The superior mesenteric artery (SMA) angiogram shows about a 7.8 mm x 3 mrm aneurysm (arrow) in the segment 6 branch of the right hepatic artery arising from the SMA, and there is extravasation of the cortrast material (arrowhead). B. The SMA angiogram obtained after embolization of the segmert 6 branch of the right hepatic artery with microcoils (arrow) shows that the aneurysm is completely occluded and the extravasation of the contrast materials is no longer seen. There is an incidentally migrated coil (arrowshead) in the segment 7 branch of the right hepatic artery.

Contrast enhanced abdomen CT obtained 2 months later shows that the hepatic subcapsular hematoma (arrow) is markedly decreased, and the embolized microcoils (arrowhead) are seen in segment 6 of the liver.

Behcet's disease is a multisystem disorder characterized by recurrent aphthous stomatitis, genital ulceration and relapsing uveitis. It most often affects men between the ages of 20 and 40 years. The disease is most prevalent in the Mediterranean region, Middle East, and Far East. The etiology of Behcet's disease is unclear (1, 2). Cardiovascular involvement appears in only 7%~29% of patients with Behcet's disease (1, 2). Vascular involvement of Behcet's disease manifests as arterial occlusion, aneurysm, venous occlusion and varices (1, 2). In vascular involvement, arterial lesion is less frequent than venous lesion and the arterial lesions accounts for only 12% of vascular complications in Behcet's disease. The arterial lesion usually develops in the aorta and pulmonary artery as well as in their major branches and an aneurysm is in 65% of patients and an occlusion in 35% (1, 2). Histologically, the vascular manifestation is a vasculitis of the vasa vasorum of the large arteries and veins that causes wall compromise, thrombosis, obstruction and aneurysm formation (3). Perforation of the arterial wall due to obliterative endarteritis of the vasa vasorum may result in aneurysm formation or rupture (3). The most common site of aneurysm formation is the abdominal aorta, and this is followed by the pulmonary, femoral, subclavian, popliteal, common carotid, coronary, brachial, ulnar, common iliac, external iliac, tibial, renal, cerebral, axillary and splenic arteries (1, 2). Aneurysm of the visceral arteries has been rarely reported on (4, 5). There is one report of a common hepatic artery aneurysm having a fistulous communication with the superior mesenteric vein in a patient with Behcet's disease (5). However, an intrahepatic artery aneurysm has not been reported in patients with Behcet's disease. Hepatic artery aneurysms constitute 20% of all visceral artery aneurysms and approximately 20% of hepatic aneurysms are intrahepatic. They are caused by atherosclerosis, vasculitis, septic emboli, iatrogenic injury or trauma (6). In this case, the patient had no history of recent or remote trauma. Therefore, vasculitis from underlying Bechet's disease is the most likely cause of the hepatic artery aneurysm. Spontaneous rupture of an arterial aneurysm is most common cause of mortality in patients with Behcet's disease (1, 2), and fast, aggressive treatment for this malady is absolutely necessary. However, the walls of the involved vessels or aneurysms in Behcet's disease are too fragile to repair. Therefore, the surgical repair of Behcet aneurysms is often unsuccessful and this treatment may result in new aneurysm and graft occlusion (7). Endovascular treatment is a reasonable alternative to prevent complications or recurrences after surgical repair. Endovascular treatments such as stent-graft or transcatheter arterial embolization have recently been reported on and they are considered to be safe and effective for the aortic and arterial aneurysms seen in Behcet's disease (8, 9). We successfully treated an intrahepatic artery aneurysm with transcatheter arterial coil embolization in a patient with Behcet's disease.

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