DOI: 10.1148/radiol.2203001418
(Radiology. 2001;220:745-750.)
© RSNA, 2001
Vascular and Interventional Radiology |
Aortic and Arterial Aneurysms in Behçet Disease: Management with Stent-Grafts—Initial Experience1
Jae Hyung Park, MD,
Jin Wook Chung, MD,
Joon Hee Joh, MD,
Soon Young Song, MD,
Sang June Shin, MD,
Kyu Sik Chung, MD,
Do Yun Lee, MD,
Jong Yoon Won, MD and
Sang Joon Kim, MD
1 From the Departments of Radiology (J.H.P., J.W.C., J.H.J., S.Y.S., S.J.S.) and Surgery (S.J.K.), Seoul National University College of Medicine, 28 Yongon-dong, Chongno-gu, Seoul 110-744, Korea; Department of Radiology, Medical College of Kosin University, Pusan, Korea (K.S.C.); and Department of Diagnostic Radiology, Yonsei University College of Medicine, Seoul, Korea (D.Y.L., J.Y.W.). Received August 18, 2000; revision requested October 18; final revision received February 21, 2001; accepted March 2. Supported by grant HMP-98-G-2-043 of the ’98 Highly Advanced National Projects on the Development of Biomedical Engineering and Technology, Ministry of Health and Welfare, Republic of Korea. Address correspondence to J.H.P. (e-mail: parkjh@radcom.snu.ac.kr).
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ABSTRACT
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PURPOSE: To assess the application of stent-grafts in the management of aortic and arterial aneurysms in patients with Behçet disease.
MATERIALS AND METHODS: Nine aneurysms in seven patients were managed with various types of stent-grafts. Diagnoses were based on clinical findings. The aortic aneurysms (n = 3) were thoracic (n = 1), suprarenal (n = 1), or infrarenal (n = 1). The arterial aneurysms (n = 6) were in the right and left subclavian (n = 2), right common carotid (n = 2), right brachiocephalic (n = 1), or left common iliac arteries (n = 1). A tandem connection of Gianturco stent covered with polytetrafluorethylene was placed in three aneurysms, and a balloon-expandable stent was placed in six.
RESULTS: The stent-graft was successfully placed in all patients. Immediate follow-up angiography revealed complete exclusion of the aneurysm in all cases. Follow-up computed tomography performed 3 days to 2 weeks later revealed complete exclusion and thrombosis of the aneurysm and patency of the stent-graft in six patients. In one patient, total occlusion of the artery with a stent occurred due to flow disturbance caused by double lesions. During follow-up (range, 6–59 months; mean, 28 months), the aneurysm resolved and completely regressed in four patients. A recurrent aneurysm at the distal margin of previously inserted stent-graft was successfully managed with an additional stent-graft.
CONCLUSION: The findings in this initial experience suggest that stent-graft insertion may be a safe and effective alternative to surgical treatment of aortic and arterial aneurysms in patients with Behçet disease.
Index terms: Aneurysm, 9*.6292 • Aneurysm, CT, 9*.1291, 9*.12916 • Behçet disease, 9*.1268 • Stents and prostheses, 9*.1268
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INTRODUCTION
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Behçet disease is a multisystemic disorder characterized by recurrent orogenital ulcers, ocular manifestations, and skin lesions. Cardiovascular involvement appears in only 7%–29% of patients. However, it is the most common cause of mortality in patients with Behçet disease. In vascular involvement, a venous lesion is more frequent, and arterial manifestations are less frequent, accounting for only 12% of vascular complications in Behçet disease. The arterial lesion, which develops in the aorta and pulmonary artery, as well as in their major branches, is an aneurysm in 65% of patients and an occlusion in 35% (1,2).
Previously, open surgical repair was the definitive treatment for vascular lesions, such as aneurysms, in patients with Behçet disease (3–6). However, the success rate of surgical management has not been high because a false aneurysm often occurs after surgical repair (5,7). To avoid surgical complications, endovascular methods, such as insertion of a stent-graft, have been recommended because they are less invasive (8,9). Recently, stent-graft placement has been indicated for the treatment of thoracic or abdominal aortic aneurysms in patients with a high surgical risk (10–14). To our knowledge, only four case reports (9,10,15,16) about the application of stent-grafts in patients with Behçet disease exist in the literature, and two of the four are from our institution.
The purpose of our study was to assess the use of stent-grafts for the management of aneurysms in patients with Behçet disease.
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MATERIALS AND METHODS
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Various types of stent-grafts were applied in nine aneurysms in seven patients (five men, two women; mean age, 37 years; age range, 32–45 years) with Behçet disease in our two hospitals. Data from one (patient 1) of the seven patients were reported in an article about the management of infrarenal aortic aneurysm with a stent-graft (15). The diagnosis of Behçet disease in all patients was made on the basis of clinical and laboratory findings. All patients except one satisfied the international diagnostic criteria for Behçet disease. Patient 6, who had bilateral neck masses due to arterial aneurysms, reported hip joint pain in addition to an evident orogenital ulcer. The diagnosis was made clinically by excluding other possibilities. The diagnostic criteria were the presence of an oral ulceration and any two of the following: genital ulceration, typically defined eye lesions, typically defined skin lesions, or a positive pathergy test result (17).
In all cases, the location, size, and shape of the nine aneurysms were confirmed with computed tomography (CT) or CT angiography and digital subtraction angiography before the procedure. The aneurysms were aortic (n = 3) or arterial (n = 6). The aortic aneurysms were thoracic (n = 1), suprarenal (n = 1), or infrarenal (n = 1). The arterial aneurysms were in the right and left subclavian (n = 2), right common carotid (n = 2), brachiocephalic (n = 1), or left common iliac arteries (n = 1).
The stent-graft was individually designed after preprocedural CT. The diameter of the stent-graft was 10%–15% larger than the measured diameter of the aorta, proximal and distal to the lesion. The length of the stent-graft was sufficient to cover 2 cm proximal and distal to the margin of the aneurysm. The stent-graft was a tandem connection of a Gianturco stent covered with polytetrafluorethylene (Tae Woong Medical, Seoul, Korea) in three patients with an aortic or left iliac arterial aneurysm and a balloon-expandable stent (Jostent-graft; Jomed, Rangendingen, Germany) in five cases of an arterial aneurysm and in one of a distal abdominal aortic aneurysm. Selection of the stent-graft for the particular patients depended on the availability of a stent-graft of proper size at the time of the procedure. The aortic stent-graft of large diameter was individually designed after preprocedural CT evaluation.
After angiographic evaluation, the introduction system, 12–24 F in outer diameter, and the stent-graft were inserted through the opening of the arteriotomy; the procedure was performed in the angiographic suite. The route of introduction involved a right or left femoral arteriotomy in six patients and a right brachial arterial puncture in one patient. Immediately before the insertion of the stent-graft, heparin 5,000–10,000 IU (Choongwae, Seoul, Korea) was intravenously administered for anticoagulation. All patients were treated with antibiotics on the day of the procedure and for 5 days after the procedure.
Follow-up clinical evaluation was performed in all patients, and follow-up CT angiography was performed 3 days to 2 weeks later and then every 6 months in all patients except patient 4. In two cases, digital subtraction angiography was performed after 1 or 6 months to confirm the exclusion of aneurysm and patency of the stent-graft. Two authors (P.J.H., C.J.H.) retrospectively reviewed the images obtained before and after the placement of the stent-graft. The images were evaluated for aneurysm shape, presence of thrombosis, location of aneurysm, wall thickness of the aneurysm and adjacent normal aorta, vessel occlusion, compression of the parent artery, type of stent-graft, success of the placement of the stent-graft, complications, and outcome.
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RESULTS
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CT findings of arterial Behçet disease included partially thrombosed irregular, lobulated, and eccentric aneurysms of the aorta or its major branches in the arterial phase (Figs 1 –3). The wall of the aneurysm was thin and well delineated, with an enhancing rim in a delayed phase in all cases (Figs 1a, 3c). In these cases, no evidence of mural thickening or enhancement in the other parts of the intact aorta existed. Angiography revealed an eccentric and lobulated aneurysm of the aorta or major arterial branches, with a variably sized aneurysm neck (Figs 2c, 3b; Table 1). The celiac and superior mesenteric arteries were occluded in two patients with an aortic aneurysm. The inferior mesenteric artery was the only mesenteric branch supplying the entire gastroenteric system in these patients. In four cases of a large eccentric aneurysm, the aneurysm compressed the parent artery.
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Figure 1a. Transverse CT angiograms in patient 1 with an infrarenal aortic aneurysm. (The initial stent-graft procedure performed in the same patient was previously reported [15].) (a) The aneurysm (arrowhead) is successfully excluded by a fenestrated-type stent-graft (arrow) and is completely filled with thrombus 2 weeks after insertion of the stent-graft. (b) The aneurysm in the infrarenal aorta has completely regressed after 53 months.
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Figure 1b. Transverse CT angiograms in patient 1 with an infrarenal aortic aneurysm. (The initial stent-graft procedure performed in the same patient was previously reported [15].) (a) The aneurysm (arrowhead) is successfully excluded by a fenestrated-type stent-graft (arrow) and is completely filled with thrombus 2 weeks after insertion of the stent-graft. (b) The aneurysm in the infrarenal aorta has completely regressed after 53 months.
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Figure 2a. Images in patient 1 with a recurrent aneurysm at the margin of the stent-graft. (a) Transverse CT scan obtained at 10-month follow-up reveals an eccentric aneurysm (arrows) at the origin of the left common iliac artery, at the distal margin of the stent-graft that was placed for an infrarenal aortic aneurysm. (b) Frontal CT angiogram, shaded-surface display, reveals the recurrent aneurysm (arrows) at the distal margin of the stent-graft. (c) Frontal abdominal aortogram shows the recurrent aneurysm (arrow) at the origin of the left common iliac artery. (d) Frontal abdominal aortogram shows that the recurrent aneurysm has almost disappeared after insertion of a second stent-graft (arrows). (e) Transverse follow-up CT scan obtained after 43 months reveals complete resolution of the aneurysm.
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Figure 2b. Images in patient 1 with a recurrent aneurysm at the margin of the stent-graft. (a) Transverse CT scan obtained at 10-month follow-up reveals an eccentric aneurysm (arrows) at the origin of the left common iliac artery, at the distal margin of the stent-graft that was placed for an infrarenal aortic aneurysm. (b) Frontal CT angiogram, shaded-surface display, reveals the recurrent aneurysm (arrows) at the distal margin of the stent-graft. (c) Frontal abdominal aortogram shows the recurrent aneurysm (arrow) at the origin of the left common iliac artery. (d) Frontal abdominal aortogram shows that the recurrent aneurysm has almost disappeared after insertion of a second stent-graft (arrows). (e) Transverse follow-up CT scan obtained after 43 months reveals complete resolution of the aneurysm.
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Figure 2c. Images in patient 1 with a recurrent aneurysm at the margin of the stent-graft. (a) Transverse CT scan obtained at 10-month follow-up reveals an eccentric aneurysm (arrows) at the origin of the left common iliac artery, at the distal margin of the stent-graft that was placed for an infrarenal aortic aneurysm. (b) Frontal CT angiogram, shaded-surface display, reveals the recurrent aneurysm (arrows) at the distal margin of the stent-graft. (c) Frontal abdominal aortogram shows the recurrent aneurysm (arrow) at the origin of the left common iliac artery. (d) Frontal abdominal aortogram shows that the recurrent aneurysm has almost disappeared after insertion of a second stent-graft (arrows). (e) Transverse follow-up CT scan obtained after 43 months reveals complete resolution of the aneurysm.
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Figure 2d. Images in patient 1 with a recurrent aneurysm at the margin of the stent-graft. (a) Transverse CT scan obtained at 10-month follow-up reveals an eccentric aneurysm (arrows) at the origin of the left common iliac artery, at the distal margin of the stent-graft that was placed for an infrarenal aortic aneurysm. (b) Frontal CT angiogram, shaded-surface display, reveals the recurrent aneurysm (arrows) at the distal margin of the stent-graft. (c) Frontal abdominal aortogram shows the recurrent aneurysm (arrow) at the origin of the left common iliac artery. (d) Frontal abdominal aortogram shows that the recurrent aneurysm has almost disappeared after insertion of a second stent-graft (arrows). (e) Transverse follow-up CT scan obtained after 43 months reveals complete resolution of the aneurysm.
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Figure 2e. Images in patient 1 with a recurrent aneurysm at the margin of the stent-graft. (a) Transverse CT scan obtained at 10-month follow-up reveals an eccentric aneurysm (arrows) at the origin of the left common iliac artery, at the distal margin of the stent-graft that was placed for an infrarenal aortic aneurysm. (b) Frontal CT angiogram, shaded-surface display, reveals the recurrent aneurysm (arrows) at the distal margin of the stent-graft. (c) Frontal abdominal aortogram shows the recurrent aneurysm (arrow) at the origin of the left common iliac artery. (d) Frontal abdominal aortogram shows that the recurrent aneurysm has almost disappeared after insertion of a second stent-graft (arrows). (e) Transverse follow-up CT scan obtained after 43 months reveals complete resolution of the aneurysm.
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Figure 3a. Images in patient 2 with a suprarenal aortic aneurysm. (a) Transverse arterial phase CT scan shows an eccentric aneurysm (arrows) with partial thrombosis at the suprarenal aorta. The celiac and superior mesenteric arteries are occluded. (b) Frontal abdominal aortogram reveals an irregular lobulated aneurysm (arrows) of the suprarenal aorta. (c) Transverse venous phase CT scan shows complete thrombosis of the aneurysm with an enhancing capsule (arrow) 5 days after insertion of the stent-graft. (d) Transverse follow-up arterial phase CT scan shows complete regression of the aneurysm after 4 months.
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Figure 3b. Images in patient 2 with a suprarenal aortic aneurysm. (a) Transverse arterial phase CT scan shows an eccentric aneurysm (arrows) with partial thrombosis at the suprarenal aorta. The celiac and superior mesenteric arteries are occluded. (b) Frontal abdominal aortogram reveals an irregular lobulated aneurysm (arrows) of the suprarenal aorta. (c) Transverse venous phase CT scan shows complete thrombosis of the aneurysm with an enhancing capsule (arrow) 5 days after insertion of the stent-graft. (d) Transverse follow-up arterial phase CT scan shows complete regression of the aneurysm after 4 months.
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Figure 3c. Images in patient 2 with a suprarenal aortic aneurysm. (a) Transverse arterial phase CT scan shows an eccentric aneurysm (arrows) with partial thrombosis at the suprarenal aorta. The celiac and superior mesenteric arteries are occluded. (b) Frontal abdominal aortogram reveals an irregular lobulated aneurysm (arrows) of the suprarenal aorta. (c) Transverse venous phase CT scan shows complete thrombosis of the aneurysm with an enhancing capsule (arrow) 5 days after insertion of the stent-graft. (d) Transverse follow-up arterial phase CT scan shows complete regression of the aneurysm after 4 months.
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Figure 3d. Images in patient 2 with a suprarenal aortic aneurysm. (a) Transverse arterial phase CT scan shows an eccentric aneurysm (arrows) with partial thrombosis at the suprarenal aorta. The celiac and superior mesenteric arteries are occluded. (b) Frontal abdominal aortogram reveals an irregular lobulated aneurysm (arrows) of the suprarenal aorta. (c) Transverse venous phase CT scan shows complete thrombosis of the aneurysm with an enhancing capsule (arrow) 5 days after insertion of the stent-graft. (d) Transverse follow-up arterial phase CT scan shows complete regression of the aneurysm after 4 months.
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The stent-graft was tubular in all arterial aneurysms except one, in which a fenestrated and tapered type was used for the aneurysm at the origin of the left iliac artery. The stent-graft was tubular or tapered in the three aortic aneurysms. The size of the stent-graft ranged from 10 to 30 mm in diameter and from 38 to 90 mm in length for aortic aneurysms and from 5 to 13 mm in diameter and from 28 to 60 mm in length for arterial aneurysms. Angiograms obtained immediately after the procedure proved that the stent-graft was successfully placed in all cases, with an initial success rate of 100%. In the five aneurysms of major arterial branches, a balloon-expandable and tubular stent-graft (Jostent-graft; Jomed) was successfully placed in all cases.
The outcomes of the stent-graft and the follow-up results are summarized in Table 2. CT performed after 3 days to 2 weeks revealed the successful exclusion of the aneurysm with the stent-graft and complete thrombosis of the aneurysm in all cases (Figs 1–3). As a complication of the procedure, a high fever developed in patient 1 for 2 days and subsided with only symptomatic treatment. There was no evidence of organisms in the blood culture of the patient with postprocedural fever.
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TABLE 2. Details of Stent-Graft Application and Follow-up for 10 Aortic or Arterial Aneurysms in Seven Patients with Behçet Disease
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Follow-up for the seven patients was as long as 59 months (range, 6–59 months; mean, 28 months ± 20 [SD]). During this period, CT was performed every 6 months after the initial CT evaluation in all patients except one (patient 4). At CT follow-up, the patency of the stent-graft was well maintained in six (86%) patients. In four patients, the aneurysm resolved and completely disappeared on the follow-up CT image obtained more than 3 months after stent-graft placement (Figs 1–3). A total occlusion of the artery with a stent occurred in patient 4, who had two aneurysms of the right common carotid artery that were most likely due to flow disturbance caused by double lesions in a vessel of small diameter. In patient 1, who had an infrarenal abdominal aortic aneurysm and patent inferior mesenteric artery as the only feeder artery for the gastrointestinal tract, a fenestrated stent-graft was inserted first to preserve the inferior mesenteric artery, as previously reported (15) (Fig 1). After 10 months, a recurrent aneurysm apparently developed at the distal margin of the stent-graft. However, this was managed with the application of an additional stent-graft. The second procedure performed to cover the recurrent aneurysm was successful. This procedure involved a tapered and fenestrated-type stent-graft; no evidence of further recurrence was found during 49-month follow-up (Fig 2e). The two aneurysms completely disappeared on the follow-up CT image obtained after 43 months; this finding suggested complete regression of the aneurysm.
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DISCUSSION
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Aneurysm is more frequent than occlusion with arterial involvement in Behçet disease. The most common site of aneurysm formation is the abdominal aorta, followed by the femoral and pulmonary arteries. In addition, abdominal aortic aneurysms are more frequent than the thoracic variety of aortic aneurysm, as was seen in our study (1).
The leading cause of death in patients with Behçet disease is a rupture of a large aortic or arterial aneurysm (1,2). At histologic analysis, aortitis is seen in both active and scar stages. Active aortitis leads to the destruction of the media and fibrosis, predisposing the patient to saccular aneurysm (18). Perforation of the arterial wall due to obliterative endarteritis of the vasa vasorum may result in aneurysm formation or rupture (9).
Many attempts have been made to surgically manage these serious arterial lesions. Surgery is available for resecting lesions and replacing grafts. Placement of a polytetrafluorethylene graft is suggested as the treatment of choice in reconstruction. However, there are several reports (3–5) concerning recurrence following surgical management in about half of the cases. Okada et al (7) reported their surgical experiences in eight cases of Behçet disease involving the ascending aorta. The mean follow-up for the eight patients was 6.8 years. Four (50%) required a second operation, and two of the four underwent a third operation due to recurrence. The interval to recurrence was variable, ranging from 10 months to 8 years. Sasaki et al (5) reported two recurrences in four patients with peripheral arterial aneurysms due to Behçet disease. The mean follow-up was 7.5 years. The interval to the recurrence was 7 days and 2 months in the two patients. To suppress possible exacerbation after surgery, adjunct medical therapy may be helpful (6).
To prevent complications from surgical repair, endovascular insertion of a stent-graft is a reasonable alternative. Recently, the stent-graft has been indicated for thoracic or abdominal aortic aneurysms—including ruptured aneurysm, peripheral arterial aneurysm, and arteriovenous fistula—when a high risk exists with surgical treatment (10–13). Presently, several types of stent-grafts are commercially available. However, each stent-graft should be custom tailored after accurate delineation and measurement of the aneurysm and parent artery with CT. Although surgical arteriotomy is a percutaneous technique, it is necessary to preserve the artery after the procedure. The administration of heparin is recommended immediately before insertion of the delivery system for the stent-graft.
The technical success rate of stent-graft insertion is higher than 90%; successful insertion results in the exclusion of the aneurysm and complete thrombosis. In cases of abdominal aortic aneurysm with successful exclusion of the aneurysm and complete thrombosis, the size of the aneurysm decreases after 12 months in about half of the cases: 43.3% with the tubular type (n = 97) and 50% with bifurcated endografts (n = 122) (19). In our series, aneurysm size rapidly decreased after successful exclusion and complete thrombosis of the aneurysm. In those cases with a follow-up longer than 3 months, the aneurysm decreased substantially and disappeared; this finding suggested complete resolution of the aneurysm.
As for the clinical application of the stent-graft in patients with Behçet disease, a fenestrated stent-graft was applied in a patient (patient 1) with abdominal aortic aneurysm (15). Ten months thereafter, placement of a second stent-graft was necessary to treat a recurrent aneurysm at the distal margin of the previously inserted stent-graft. When the reported interval to recurrence of a false aneurysm is considered, the recurrence after 10 months in patient 1 may have been the development of a new false aneurysm induced by a minor injury at the site of stent-graft as a natural progression of the disease. However, the patient had been followed up for longer than 4 years to prove that the two aneurysms disappeared completely. Thus, recurrence is evidently possible in Behçet disease even after treatment with a stent-graft. However, the recurrence may be controlled with the repeated use of stent-grafts. As another complication of stent-graft insertion, one case (in patient 4) of occlusion occurred at the right common carotid artery in two stent-grafts. The double lesions and smaller inner diameter of the carotid artery may have predisposed the artery to occlusion.
Vasseur et al (9) reported a case of Behçet disease in which they successfully treated a false aneurysm in the infrarenal abdominal aorta with a bifurcated type of stent-graft (Vanguard; Boston Scientific, Boston, Mass). The aneurysm regressed completely after 6-month follow-up with CT and duplex scanning. In our series with long-term follow-up of as long as 59 months, the aneurysm resolved and completely regressed in four patients. We think that complete regression of an aneurysm can be expected after about 3 months if the stent-graft successfully excludes the aneurysm in patients with Behçet disease. Because the aneurysm has no true vascular wall and because it has a pseudocapsule composed of surrounding connective tissue and thrombi, the aneurysm may rapidly decrease in size and completely regress after successful exclusion, due to resolution of the thrombi and regression of the pseudocapsule.
Bonnotte et al (8) reported a case of a false aneurysm in the internal carotid artery in Behçet disease that was successfully managed with a bare stent and coil embolization. A metallic stent without graft coverage may be easily applied in smaller vessels to result in a gradual exclusion of the aneurysm; however, it may not replace the stent-graft in large aneurysms of the aorta (20).
In summary, the findings of our initial experience suggest that stent-graft insertion may be a safe and effective alternative to surgical treatment for aortic and arterial aneurysms in patients with Behçet disease and may result in complete regression of aneurysms at long-term follow-up. Further investigation with longer-term follow-up is strongly recommended to confirm that the stent-graft is not only an alternative treatment to surgery but also a definitive treatment for aortic and arterial aneurysms in patients with Behçet disease.
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FOOTNOTES
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2 9*. Vascular system, location unspecified 
Author contributions: Guarantor of integrity of entire study, J.H.P.; study concepts, J.H.P., J.W.C., D.Y.L.; study design, J.H.J., S.Y.S., S.J.S., K.S.C., J.Y.W.; literature research, J.H.J.; clinical studies, J.W.C., S.J.S., K.S.C., D.Y.L., J.Y.W., S.Y.S.; data acquisition, J.H.P., J.H.J., D.Y.L., J.Y.W.; data analysis/interpretation, J.H.P., J.W.C., J.H.J.; manuscript preparation, J.H.J., S.Y.S.; manuscript definition of intellectual content, J.H.P., J.W.C., J.H.J., S.J.K.; manuscript editing, J.H.P., J.H.J.; manuscript revision/review, J.W.C., J.H.J.; manuscript final version approval, J.H.P.
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N. Y. Jung, S. K. Kim, E. C. Chung, H. Park, and Y. K. Cho
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TOP
ABSTRACT
INTRODUCTION
