Jae Hyung Park, MDJin Wook Chung, MDJoon Hee Joh, MDSoon Young Song, MDSang June Shin, MDKyu Sik Chung, MDDo Yun Lee, MDJong Yoon Won, MDSang Joon Kim, MD

Index terms:Aneurysm, 9p.6292

Aneurysm, CT, 9p.1291, 9p.12916Behcet disease, 9p.1268Stents and prostheses, 9p.1268

Published online: August 21, 200110.1148/radiol.2203001418

Radiology 2001; 220:745750

1 From the Departments of Radiology(J.H.P., J.W.C., J.H.J., S.Y.S., S.J.S.) andSurgery (S.J.K.), Seoul National Univer-sity College of Medicine, 28 Yongon-dong, Chongno-gu, Seoul 110-744, Ko-rea; Department of Radiology, MedicalCollege of Kosin University, Pusan, Ko-rea (K.S.C.); and Department of Diag-nostic Radiology, Yonsei University Col-lege of Medicine, Seoul, Korea (D.Y.L.,J.Y.W.). Received August 18, 2000; revi-sion requested October 18; final revi-sion received February 21, 2001; ac-cepted March 2. Supported by grantHMP-98-G-2-043 of the 98 Highly Ad-vanced National Projects on the Devel-opment of Biomedical Engineering andTechnology, Ministry of Health andWelfare, Republic of Korea. Addresscorrespondence to J.H.P. (e-mail:parkjh@radcom.snu.ac.kr).

2 9p. Vascular system, location unspeci-fied RSNA, 2001

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 intellectualcontent, J.H.P., J.W.C., J.H.J., S.J.K.;manuscript editing, J.H.P., J.H.J.; manu-script revision/review, J.W.C., J.H.J.;manuscript final version approval, J.H.P.

Aortic and Arterial Aneurysmsin Behcet Disease: Managementwith Stent-GraftsInitialExperience1

PURPOSE: To assess the application of stent-grafts in the management of aortic andarterial aneurysms in patients with Behcet disease.

MATERIALS AND METHODS: Nine aneurysms in seven patients were managedwith various types of stent-grafts. Diagnoses were based on clinical findings. Theaortic aneurysms (n 5 3) were thoracic (n 5 1), suprarenal (n 5 1), or infrarenal(n 5 1). The arterial aneurysms (n 5 6) were in the right and left subclavian (n 5 2),right common carotid (n 5 2), right brachiocephalic (n 5 1), or left common iliacarteries (n 5 1). A tandem connection of Gianturco stent covered with polytetraflu-orethylene was placed in three aneurysms, and a balloon-expandable stent wasplaced in six.

RESULTS: The stent-graft was successfully placed in all patients. Immediate fol-low-up angiography revealed complete exclusion of the aneurysm in all cases.Follow-up computed tomography performed 3 days to 2 weeks later revealedcomplete exclusion and thrombosis of the aneurysm and patency of the stent-graftin six patients. In one patient, total occlusion of the artery with a stent occurred dueto flow disturbance caused by double lesions. During follow-up (range, 659months; mean, 28 months), the aneurysm resolved and completely regressed infour patients. A recurrent aneurysm at the distal margin of previously insertedstent-graft was successfully managed with an additional stent-graft.

CONCLUSION: The findings in this initial experience suggest that stent-graft inser-tion may be a safe and effective alternative to surgical treatment of aortic and arterialaneurysms in patients with Behcet disease.

Behcet disease is a multisystemic disorder characterized by recurrent orogenital ulcers,ocular manifestations, and skin lesions. Cardiovascular involvement appears in only7%29% of patients. However, it is the most common cause of mortality in patients withBehcet disease. In vascular involvement, a venous lesion is more frequent, and arterialmanifestations are less frequent, accounting for only 12% of vascular complications inBehcet disease. The arterial lesion, which develops in the aorta and pulmonary artery, aswell 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 asaneurysms, in patients with Behcet disease (36). However, the success rate of surgicalmanagement 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 astent-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 aorticaneurysms in patients with a high surgical risk (1014). To our knowledge, only four casereports (9,10,15,16) about the application of stent-grafts in patients with Behcet diseaseexist 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 ofaneurysms in patients with Behcet disease.

745

MATERIALS AND METHODS

Various types of stent-grafts were appliedin nine aneurysms in seven patients (fivemen, two women; mean age, 37 years;age range, 3245 years) with Behcet dis-ease in our two hospitals. Data from one(patient 1) of the seven patients were re-ported in an article about the manage-ment of infrarenal aortic aneurysm witha stent-graft (15). The diagnosis of Behcetdisease in all patients was made on thebasis of clinical and laboratory findings.All patients except one satisfied the inter-national diagnostic criteria for Behcetdisease. Patient 6, who had bilateral neckmasses due to arterial aneurysms, re-ported hip joint pain in addition to anevident orogenital ulcer. The diagnosiswas made clinically by excluding otherpossibilities. The diagnostic criteria werethe presence of an oral ulceration andany two of the following: genital ulcer-ation, typically defined eye lesions, typi-cally defined skin lesions, or a positivepathergy test result (17).

In all cases, the location, size, andshape of the nine aneurysms were con-firmed with computed tomography (CT)or CT angiography and digital subtrac-tion angiography before the procedure.The aneurysms were aortic (n 5 3) orarterial (n 5 6). The aortic aneurysmswere thoracic (n 5 1), suprarenal (n 5 1),or infrarenal (n 5 1). The arterial aneu-rysms were in the right and left subcla-vian (n 5 2), right common carotid (n 52), brachiocephalic (n 5 1), or left com-mon iliac arteries (n 5 1).

The stent-graft was individually de-

signed after preprocedural CT. The diam-eter of the stent-graft was 10%15%larger than the measured diameter of theaorta, proximal and distal to the lesion.The length of the stent-graft was suffi-cient to cover 2 cm proximal and distal tothe margin of the aneurysm. The stent-graft was a tandem connection of a Gi-anturco stent covered with polytetraflu-orethylene (Tae Woong Medical, Seoul,Korea) in three patients with an aortic orleft iliac arterial aneurysm and a balloon-expandable stent (Jostent-graft; Jomed,Rangendingen, Germany) in five cases ofan arterial aneurysm and in one of a dis-tal abdominal aortic aneurysm. Selectionof the stent-graft for the particular pa-tients depended on the availability of astent-graft of proper size at the time ofthe procedure. The aortic stent-graft oflarge diameter was individually designedafter preprocedural CT evaluation.

After angiographic evaluation, the in-troduction system, 1224 F in outer di-ameter, and the stent-graft were insertedthrough the opening of the arteriotomy;the procedure was performed in the an-giographic suite. The route of introduc-tion involved a right or left femoral arte-riotomy in six patients and a rightbrachial arterial puncture in one patient.Immediately before the insertion of thestent-graft, heparin 5,00010,000 IU(Choongwae, Seoul, Korea) was intrave-nously administered for anticoagulation.All patients were treated with antibioticson the day of the procedure and for 5days after the procedure.

Follow-up clinical evaluation was per-formed in all patients, and follow-up CT

angiography was performed 3 days to 2weeks later and then every 6 months inall patients except patient 4. In two cases,digital subtraction angiography was per-formed after 1 or 6 months to confirmthe exclusion of aneurysm and patencyof the stent-graft. Two authors (P.J.H.,C.J.H.) retrospectively reviewed the im-ages obtained before and after the place-ment of the stent-graft. The images wereevaluated for aneurysm shape, presenceof thrombosis, location of aneurysm,wall thickness of the aneurysm and adja-cent normal aorta, vessel occlusion, com-pression of the parent artery, type of stent-graft, success of the placement of the stent-graft, complications, and outcome.

RESULTS

CT findings of arterial Behcet disease in-cluded partially thrombosed irregular,lobulated, and eccentric aneurysms ofthe aorta or its major branches in thearterial phase (Figs 13). The wall of theaneurysm was thin and well delineated,with an enhancing rim in a delayedphase in all cases (Figs 1a, 3c). In thesecases, no evidence of mural thickening orenhancement in the other parts of theintact aorta existed. Angiography re-vealed an eccentric and lobulated aneu-rysm of the aorta or major arterialbranches, with a variably sized aneurysmneck (Figs 2c, 3b; Table 1). The celiac andsuperior mesenteric arteries were occludedin two patients with an aortic aneurysm.The inferior mesenteric artery was the onlymesenteric branch supplying the entire

Figure 1. Transverse CT angiograms in patient 1 with an infrarenal aortic aneurysm. (The initial stent-graft procedure performed in the samepatient was previously reported [15].) (a) The aneurysm (arrowhead) is successfully excluded by a fenestrated-type stent-graft (arrow) and iscompletely filled with thrombus 2 weeks after insertion of the stent-graft. (b) The aneurysm in the infrarenal aorta has completely regressed after53 months.

746 z Radiology z September 2001 Park et al

gastroenteric system in these patients. Infour cases of a large eccentric aneurysm,the aneurysm compressed the parent ar-tery.

The stent-graft was tubular in all arterialaneurysms except one, in which a fenes-trated and tapered type was used for theaneurysm at the origin of the left iliac ar-tery. The stent-graft was tubular or taperedin the three aortic aneurysms. The size ofthe stent-graft ranged from 10 to 30 mm indiameter and from 38 to 90 mm in lengthfor aortic aneurysms and from 5 to 13 mmin diameter and from 28 to 60 mm inlength for arterial aneurysms. Angiogramsobtained immediately after the procedureproved that the stent-graft was successfullyplaced in all cases, with an initial successrate of 100%. In the five aneurysms ofmajor arterial branches, a balloon-ex-pandable and tubular stent-graft (Jostent-graft; Jomed) was successfully placed inall cases.

The outcomes of the stent-graft and thefollow-up results are summarized in Table2. CT performed after 3 days to 2 weeks

revealed the successful exclusion of the an-eurysm with the stent-graft and completethrombosis of the aneurysm in all cases(Figs 13). As a complication of the proce-dure, a high fever developed in patient 1for 2 days and subsided with only symp-tomatic treatment. There was no evidenceof organisms in the blood culture of thepatient with postprocedural fever.

Follow-up for the seven patients was aslong as 59 months (range, 659 months;mean, 28 months 6 20 [SD]). During thisperiod, CT was performed every 6 monthsafter the initial CT evaluation in all pa-tients except one (patient 4). At CT follow-up, the patency of the stent-graft was wellmaintained in six (86%) patients. In fourpatients, the aneurysm resolved and com-pletely disappeared on the follow-up CTimage obtained more than 3 months afterstent-graft placement (Figs 13). A total oc-clusion of the artery with a stent occurredin patient 4, who had two aneurysms ofthe right common carotid artery that weremost likely due to flow disturbance causedby double lesions in a vessel of small diam-

eter. In patient 1, who had an infrarenalabdominal aortic aneurysm and patent in-ferior mesenteric artery as the only feederartery for the gastrointestinal tract, a fenes-trated stent-graft was inserted first to pre-serve the inferior mesenteric artery, as pre-viously reported (15) (Fig 1). After 10months, a recurrent aneurysm apparentlydeveloped at the distal margin of the stent-graft. However, this was managed with theapplication of an additional stent-graft.The second procedure performed to coverthe recurrent aneurysm was successful.This procedure involved a tapered andfenestrated-type stent-graft; no evidence offurther recurrence was found during 49-month follow-up (Fig 2e). The two aneu-rysms completely disappeared on the fol-low-up CT image obtained after 43months; this finding suggested completeregression of the aneurysm.

DISCUSSION

Aneurysm is more frequent than occlu-sion with arterial involvement in Behcet

Figure 2. Images in patient 1 with a recurrentaneurysm at the margin of the stent-graft.(a) Transverse CT scan obtained at 10-monthfollow-up reveals an eccentric aneurysm (ar-rows) at the origin of the left common iliacartery, at the distal margin of the stent-graft thatwas placed for an infrarenal aortic aneurysm.(b) Frontal CT angiogram, shaded-surface dis-play, reveals the recurrent aneurysm (arrows) atthe distal margin of the stent-graft. (c) Frontalabdominal aortogram shows the recurrent an-eurysm (arrow) at the origin of the left commoniliac artery. (d) Frontal abdominal aortogramshows that the recurrent aneurysm has almostdisappeared after insertion of a second stent-graft (arrows). (e) Transverse follow-up CT scanobtained after 43 months reveals complete res-olution of the aneurysm.

Volume 220 z Number 3 Aortic and Arterial Aneurysms in Behcet Disease: Stent-Grafts z 747

disease. The most common site of aneu-rysm formation is the abdominal aorta,followed by the femoral and pulmonaryarteries. In addition, abdominal aorticaneurysms are more frequent than thethoracic variety of aortic aneurysm, aswas seen in our study (1).

The leading cause of death in patientswith Behcet disease is a rupture of a largeaortic or arterial aneurysm (1,2). At his-tologic analysis, aortitis is seen in bothactive and scar stages. Active aortitisleads to the destruction of the media andfibrosis, predisposing the patient to sac-cular aneurysm (18). Perforation of thearterial wall due to obliterative endarteri-tis of the vasa vasorum may result in an-eurysm formation or rupture (9).

Many attempts have been made to sur-gically manage these serious arterial le-sions. Surgery is available for resectinglesions and replacing grafts. Placement ofa polytetrafluorethylene graft is sug-gested as the treatment of choice in re-construction. However, there are severalreports (35) concerning recurrence fol-lowing surgical management in abouthalf of the cases. Okada et al (7) reportedtheir surgical experiences in eight casesof Behcet disease involving the ascendingaorta. The mean follow-up for the eightpatients was 6.8 years. Four (50%) re-quired a second operation, and two ofthe four underwent a third operation dueto recurrence. The interval to recurrencewas variable, ranging from 10 months to8 years. Sasaki et al (5) reported two re-currences in four patients with peripheralarterial aneurysms due to Behcet disease.The mean follow-up was 7.5 years. Theinterval to the recurrence was 7 days and2 months in the two patients. To sup-press possible exacerbation after surgery,adjunct medical therapy may be helpful(6).

To prevent complications from surgi-cal repair, endovascular insertion of astent-graft is a reasonable alternative. Re-cently, the stent-graft has been indicatedfor thoracic or abdominal aortic aneu-rysmsincluding ruptured aneurysm,peripheral arterial aneurysm, and arterio-venous fistulawhen a high risk existswith surgical treatment (1013). Pres-ently, several types of stent-grafts arecommercially available. However, eachstent-graft should be custom tailored af-ter accurate delineation and measure-ment of the aneurysm and parent arterywith CT. Although surgical arteriotomyis a percutaneous technique, it is neces-sary to preserve the artery after the pro-cedure. The administration of heparin isrecommended immediately before inser-

tion of the delivery system for the stent-graft.

The technical success rate of stent-graftinsertion is higher than 90%; successfulinsertion results in the exclusion of theaneurysm and complete thrombosis. Incases of abdominal aortic aneurysm withsuccessful exclusion of the aneurysm andcomplete thrombosis, the size of the an-eurysm decreases after 12 months inabout half of the cases: 43.3% with thetubular type (n 5 97) and 50% with bi-furcated endografts (n 5 122) (19). In ourseries, aneurysm size rapidly decreasedafter successful exclusion and completethrombosis of the aneurysm. In thosecases with a follow-up longer than 3months, the aneurysm decreased sub-stantially and disappeared; this findingsuggested complete resolution of the an-eurysm.

As for the clinical application of thestent-graft in patients with Behcet dis-ease, a fenestrated stent-graft was appliedin a patient (patient 1) with abdominalaortic aneurysm (15). Ten months there-after, placement of a second stent-graftwas necessary to treat a recurrent aneu-rysm at the distal margin of the previ-ously inserted stent-graft. When the re-ported interval to recurrence of a falseaneurysm is considered, the recurrenceafter 10 months in patient 1 may havebeen the development of a new false an-eurysm induced by a minor injury at thesite of stent-graft as a natural progressionof the disease. However, the patient hadbeen followed up for longer than 4 yearsto prove that the two aneurysms disap-peared completely. Thus, recurrence isevidently possible in Behcet disease evenafter treatment with a stent-graft. How-

Figure 3. Images in patient 2 with a suprarenal aortic aneurysm. (a) Transverse arterial phase CTscan shows an eccentric aneurysm (arrows) with partial thrombosis at the suprarenal aorta. Theceliac and superior mesenteric arteries are occluded. (b) Frontal abdominal aortogram reveals anirregular lobulated aneurysm (arrows) of the suprarenal aorta. (c) Transverse venous phase CTscan shows complete thrombosis of the aneurysm with an enhancing capsule (arrow) 5 days afterinsertion of the stent-graft. (d) Transverse follow-up arterial phase CT scan shows completeregression of the aneurysm after 4 months.

748 z Radiology z September 2001 Park et al

ever, the recurrence may be controlledwith the repeated use of stent-grafts. Asanother complication of stent-graft inser-tion, one case (in patient 4) of occlusionoccurred at the right common carotid ar-tery in two stent-grafts. The double le-sions and smaller inner diameter of thecarotid artery may have predisposed theartery to occlusion.

Vasseur et al (9) reported a case ofBehcet disease in which they successfullytreated a false aneurysm in the infrarenalabdominal aorta with a bifurcated type ofstent-graft (Vanguard; Boston Scientific,Boston, Mass). The aneurysm regressedcompletely after 6-month follow-up withCT and duplex scanning. In our serieswith long-term follow-up of as long as 59months, the aneurysm resolved andcompletely regressed in four patients. We

think that complete regression of an an-eurysm can be expected after about 3months if the stent-graft successfully ex-cludes the aneurysm in patients with Be-hcet disease. Because the aneurysm hasno true vascular wall and because it has apseudocapsule composed of surroundingconnective tissue and thrombi, the aneu-rysm may rapidly decrease in size andcompletely regress after successful exclu-sion, due to resolution of the thrombiand regression of the pseudocapsule.

Bonnotte et al (8) reported a case of afalse aneurysm in the internal carotid ar-tery in Behcet disease that was success-fully managed with a bare stent and coilembolization. A metallic stent withoutgraft coverage may be easily applied insmaller vessels to result in a gradual ex-clusion of the aneurysm; however, it may

not replace the stent-graft in large aneu-rysms of the aorta (20).

In summary, the findings of our initialexperience suggest that stent-graft inser-tion may be a safe and effective alternativeto surgical treatment for aortic and arterialaneurysms in patients with Behcet diseaseand may result in complete regression ofaneurysms at long-term follow-up. Furtherinvestigation with longer-term follow-up isstrongly recommended to confirm that thestent-graft is not only an alternative treat-ment to surgery but also a definitive treat-ment for aortic and arterial aneurysms inpatients with Behcet disease.

References1. Park JH, Han MC, Bettmann MA. Arterial

manifestations of Behcets disease. AJRAm J Roentgenol 1984; 143:821825.

TABLE 1Clinical Features of Seven Patients with Behcet Disease and Aortic or Arterial Aneurysm

PatientNo./Sex/Age (y) Chief Complaint Criteria* Previous Surgery Aneurysm Location

AneurysmSize (cm) Angiographic Finding

1/M/40 Back pain O, G, S, E Patch graft (11 moprior), IRAo

IRAo, recurred 2.0 Occlusion: celiac axis, both L4 arteries

1/M/40 Back pain O, G, S, E Stent-graft, IRAo Aortoiliac junction, recurred 2.0 Occlusion: celiac axis, SMA2/F/45 Back pain O, G, S, E None Suprarenal aorta 4.5 Occlusion: celiac axis, SMA3/M/37 Back pain O, G, S None Descending thoracic aorta 10.0 Compression: descending thoracic aorta4/M/32 Right neck mass O, G, S Saphenous vein graft

(3 mo prior), rightCCA

Right CCA, proximal andbifurcation, anastomotic

3.0, 4.0 Compression: right CCA

5/M/38 Right neck mass O, G, S None Right SCA 3.0 Compression: right SCA6/M/33 Bilateral neck

massesO, G, S, GI None Right BCA, left SCA 4.0, 2.5 Both CCAs patent

7/F/33 Back pain O, G, S None IRAo 2.8 Compression: distal IRAo, occlusion:right L4 artery

* E 5 eye lesion, G 5 genital ulcer, GI 5 gastrointestinal lesion, O 5 oral ulcer, S 5 skin lesion. BCA 5 brachiocephalic artery, CCA 5 common carotid artery, IRAo 5 infrarenal aorta, SCA 5 subclavian artery, SMA 5 superior mesenteric artery. The initial procedure was previously reported (15).

TABLE 2Details of Stent-Graft Application and Follow-up for 10 Aortic or Arterial Aneurysms in Seven Patients with Behcet Disease

PatientNo.

Stent-GraftFollow-up

Site* Material Type Size (mm)Duration

(mo) Study Outcome

1 IRAo G, PTFE Fenestrated, tubular 15 3 70 59 CT, angiography Recurred at distal margin1 Aorta, left iliac artery G, PTFE Fenestrated, tapered 13 3 60 49 CT, angiography Complete resolution2 Suprarenal aorta G, PTFE Tubular 22 3 75 40 CT Complete resolution3 Descending thoracic

aortaG, PTFE Tapered 30 3 90 31 CT Complete resolution

4 Right CCA, proximaland bifurcation

Both J Both tubular 5 3 40,6 3 40

6 Angiography Occlusion

5 Right SCA J Tubular 7 3 28 21 CT Complete exclusion and thrombosis6 Right BCA, left SCA Both J Both tubular Both 10 3 38 10 CT Complete resolution7 IRAo J Tubular 10 3 38 8 CT Complete exclusion and thrombosis

* BCA 5 brachiocephalic artery, CCA 5 common carotid artery, IRAo 5 infrarenal aorta, SCA 5 subclavian artery. G 5 Gianturco stent, J 5 Jostent-graft (Jomed), PTFE 5 polytetrafluoroethylene. Diameter 3 length. The initial procedure was previously reported (15).

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2. Koc Y, Gullu I, Akpek G, et al. Vascularinvolvement in Behcets disease. J Rheu-matol 1992; 19:402410.

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4. Bastounis E, Maltexos C, Gianmbouras S,Vayiopoulos G, Galas P. Arterial aneu-rysms in Behcets disease. Int Angiol1994; 13:196201.

5. Sasaki S, Yasuda K, Takigami K, Shiiya N,Matsui Y, Sakuma M. Surgical experienceswith peripheral arterial aneurysms due tovasculo-Behcets disease. J CardiovascSurg (Torino) 1998; 39:147150.

6. Barlas S. Behcets disease: an insight froma vascular surgeons point of view. ActaChir Belg 1999; 99:274281.

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8. Bonnotte B, Krause D, Fanton AL, Theron J,Chauffert B, Lorcerie B. False aneurysm ofthe internal carotid artery in Behcets dis-ease: successful combined endovasculartreatment with stent and coils (letter).Rheumatology (Oxford) 1999; 38:576577.

9. Vasseur MA, Haulon S, Beregi JP, Le Tour-neau T, Prat A, Warembourgh H. Endo-vascular treatment of abdominal aneurys-mal aortitis in Behcets disease. J VascSurg 1998; 27:974976.

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14. Schonholz C, Donnini F, Naselli G, Po-covi A, Parodi JC. Acute rupture of anaortic false aneurysm treated with a stent-graft. J Endovasc Surg 1999; 6:293296.

15. Park JH, Chung JW, Choo IW, Kim SJ, Lee

JY, Han MC. Fenestrated stent-grafts forpreserving visceral arterial branches inthe treatment of abdominal aortic aneu-rysms: preliminary experience. J Vasc In-terv Radiol 1996; 7:819823.

16. Kawaguchi S, Ishimaru S, Koizumi N,Shimazaki T, Obitsu Y, Ishikawa M. Pre-diction of spinal cord ischemia with aretrievable stent graft on endovasculartreatment for a case of thoracic aortic an-eurysm. Jpn J Thorax Cardiovasc Surg1998; 46:10471051.

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