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DOI:10.2214/AJR.05.0628
AJR 2007; 188:W400-W402
© American Roentgen Ray Society

Case Report

Endovascular Treatment for Rupture of Intrahepatic Artery Aneurysm in a Patient with Behçet's Syndrome

Na Young Jung1, Seung Kwon Kim1, Eun Chul Chung1, Haewon Park1 and Yong Kyun Cho2

1 Department of Radiology, Kangbuk Samsung Hospital, Sungkyunkwan University School of Medicine, 180, Pyung-Dong, Jongro-Ku, Seoul 110-746, South Korea.
2 Department of Internal Medicine, Kangbuk Samsung Hospital, Sungkyunkwan University School of Medicine, Seoul 110-746, South Korea.

Received April 12, 2005; accepted after revision June 24, 2005.

 
Address correspondence to S. K. Kim (radi{at}lycos.co.kr).

WEB This is a Web exclusive article.

Keywords: Behçet's syndrome • digital subtraction angiography • interventional radiology • intrahepatic artery • liver


Introduction
Top
 Introduction
Case Report
 Discussion
 References
 
Behçet's syndrome is a multisystem disorder characterized by recurrent aphthous stomatitis, genital ulceration, and relapsing uveitis. It most often affects men between 20 and 40 years old. The syndrome is most prevalent in the Mediterranean region, Middle East, and Far East. The cause of Behçet's syndrome is unclear [1, 2]. Cardiovascular involvement appears in only 7-29% of patients [1, 2]. Vascular involvement of Behçet's syndrome manifests as arterial occlusion, aneurysm, venous occlusion, and varices [1, 2].

To our knowledge, spontaneous rupture of an intrahepatic artery aneurysm has not been reported in patients with Behçet's syndrome. We describe a case of spontaneous rupture of an intrahepatic artery aneurysm in a patient with Behçet's syndrome that we successfully treated by performing transcatheter arterial coil embolization.


Case Report
Top
 Introduction
 Case Report
Discussion
 References
 
A 33-year-old man with Behçet's syndrome presented with right upper abdominal pain that he had experienced for 1 day. He was admitted to our hospital. There was no history of trauma. He was diagnosed with Behçet's syndrome 3 years before at a different hospital, and the diagnosis was confirmed by symptoms of recurrent oral and genital ulcerations and relapsing uveitis. The patient did not have fever or chills. The initial hemoglobin was 11.3 g/dL and the hematocrit was 34.1%.

Unenhanced and contrast-enhanced abdominal CT was then performed. The CT images showed a large subcapsular hematoma of mixed attenuation at the lateral and posterior aspects of the right lobe of the liver (Fig. 1A). As a result, the liver was compressed by the hematoma. There was a small high-attenuating focus within the hematoma, compatible with extravasation of the contrast material and active bleeding (Fig. 1B).


Figure 1
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  		Fig. 1A —33-year-old man with Behçet's syndrome. Contrast-enhanced abdominal CT images show large hematoma (arrows) of mixed attenuation at lateral and posterior aspects of right lobe of liver along with compression of adjacent liver. Small high-attenuating focus (arrowhead, B) is noted within hematoma, suggesting extravasation of contrast material and active bleeding.

 

Figure 2
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  		Fig. 1B —33-year-old man with Behçet's syndrome. Contrast-enhanced abdominal CT images show large hematoma (arrows) of mixed attenuation at lateral and posterior aspects of right lobe of liver along with compression of adjacent liver. Small high-attenuating focus (arrowhead, B) is noted within hematoma, suggesting extravasation of contrast material and active bleeding.

 
Angiography and embolization of the aneurysm were then planned. A 5-French Yashiro type catheter (Glidecath, Terumo) 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. 1C). The selective hepatic angiogram that was obtained using a 2.4-French microcatheter (Progreat, Terumo) clearly showed an approximately 7.8 x 3.0 mm aneurysm. The 2.4-French microcatheter was replaced with a 2-French microcatheter (Progreat) for the 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 have been due to inadequate advancement of the microcatheter to the segment 6 branch of the right hepatic artery.


Figure 3
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  		Fig. 1C —33-year-old man with Behçet's syndrome. Superior mesenteric artery (SMA) angiogram shows approximately 7.8 x 3.0 mm aneurysm (arrow) in segment 6 branch of right hepatic artery arising from SMA, and there is extravasation of contrast material (arrowhead).

 
After further advancement of the microcatheter to the segment 6 branch of the right hepatic artery, the aneurysm was successfully embolized using two 2-mm microcoils (Hilal Coil, Cook). 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 contrast material was no longer seen. The incidentally migrated coil was noted in the segment 7 branch of the right hepatic artery (Fig. 1D).


Figure 4
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  		Fig. 1D —33-year-old man with Behçet's syndrome. SMA angiogram obtained after embolization of segment 6 branch of right hepatic artery with microcoils (arrow) shows that aneurysm is completely occluded, and extravasation of contrast material is no longer seen. Incidentally migrated coil (arrowhead) is seen in segment 7 branch of right hepatic artery.

 
After 1 week, abdominal CT images 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, abdominal CT was performed again. The CT images showed the greatly decreased size of the subcapsular hematoma of the liver (Fig. 1E). Clinical follow-up at 1 year showed normal findings.


Figure 5
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  		Fig. 1E —33-year-old man with Behçet's syndrome. Contrast-enhanced abdominal CT image obtained 2 months later shows that hepatic subcapsular hematoma (arrow) is markedly decreased, and embolized microcoils (arrowhead) are seen in segment 6 of liver.

 

Discussion
Top
 Introduction
 Case Report
 Discussion
References
 
Behçet's syndrome is now recognized as a systemic disorder with mucocutaneous, ophthalmic, neurologic, cardiovascular, pulmonary, gastrointestinal, urogenital, and muscu-loskeletal involvement [1, 2]. Any artery or vein of the body may be affected, and the involvement of the vascular tree manifests itself pathologically as arterial occlusion, aneurysm, venous occlusion, and varices [1, 2]. In vascular involvement, arterial lesions are less frequent than venous lesions, and the arterial lesions account for only 12% of vascular complications in Behçet's syndrome. The arterial lesions usually develop in the aorta and pulmonary artery and in their major branches. An aneurysm is present in 65% of patients with an arterial lesion 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 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 [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 Behçet's syndrome [5]. However, an intrahepatic artery aneurysm has not been reported in patients with Behçet's syndrome.

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 Behçet's syndrome is the most likely cause of the hepatic artery aneurysm.

Spontaneous rupture of an arterial aneu-rysm is the most common cause of mortality in patients with Behçet's syndrome [1, 2], and fast, aggressive treatment for this malady is absolutely necessary. However, the walls of the involved vessels or aneurysms in Behçet's syndrome are too fragile to repair. Therefore, surgical repair of these aneurysms is often unsuccessful and may result in new aneurysm formation 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 and they are considered to be safe and effective for the aortic and arterial aneurysms seen in Behçet's syndrome [8, 9].

The migration of one microcoil may have been due to inadequate advancement of microcatheter to the segment 6 branch of the right hepatic artery. This migration could have been prevented by using a guiding catheter to obtain more secure purchase and advancement of the 5-French catheter into the SMA. No sequelae from the migrated microcoil were seen on the follow-up CT scan, which may have been due to adequate collateral arteries.

We report the successful treatment of an intrahepatic artery aneurysm with transcatheter arterial coil embolization in a patient with Behçet's syndrome.


References
Top
 Introduction
 Case Report
 Discussion
 References
 

  1. Koc Y, Gullu I, Akpek G, et al. Vascular involvement in Behcet's disease. J Rheumatol 1992;19 : 402-410[Medline]
  2. Park JH, Han MC, Bettmann MA. Arterial manifestations of Behcet's disease. AJR 1984;143 : 821-825[Abstract/Free Full Text]
  3. Matsumoto T, Uekusa T, Fukuda Y. Vasculo-Behcet's disease: a pathologic study of eight cases. Hum Pathol1991; 22:45 -51[CrossRef][Medline]
  4. Men S, Ozmen MN, Balkanci F, Boyacigil S, Akbari H. Superior mesenteric artery aneurysm in Behcet's disease. Abdom Imaging 1994; 19:333 -334[Medline]
  5. Cekirge S, Gulsun M, Oto A, Dogan R, Balkanci F, Besim A. Endovascular treatment of an unusual arterioportal fistula caused by the rupture of a giant hepatic artery aneurysm into the superior mesenteric vein in Behcet disease. J Vasc Interv Radiol2000; 11:465 -467[CrossRef][Medline]
  6. O'Driscoll D, Olliff SP, Olliff JF. Hepatic artery aneurysm. Br J Radiol 1999;72 : 1018-1025[Abstract]
  7. Sasaki S, Yasuda K, Takigami K, Shiiya N, Matsui Y, Sakuma M. Surgical experiences with peripheral arterial aneurysms due to vasculo-Behcet's disease. J Cardiovasc Surg (Torino)1998; 39:147 -150[Medline]
  8. Kasirajan K, Marek JM, Langsfeld M. Behcet's disease: endovascular management of a ruptured peripheral arterial aneurysm. J Vasc Surg 2001; 34:1127 -1129[CrossRef][Medline]
  9. Park JH, Chung JW, Joh JH, et al. Aortic and arterial aneurysms in Behcet disease: management with stent-grafts—initial experience. Radiology 2001;220 : 745-750[Abstract/Free Full Text]

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