European Journal of Vascular & Endovascular Surgery
Volume 35, Issue 2 , Pages 173-180, February 2008

Surgical Treatment Result of Abdominal Aortic Aneurysm in Behçet's Disease

  • T.-W. Kwon

      Affiliations

    • Division of Vascular Surgery, Department of Surgery, University of Ulsan College of Medicine and Asan Medical center, Seoul, Korea
    • Corresponding Author InformationCorresponding author. T.-W. Kwon, MD, PhD, Professor of Surgery, Division of Vascular Surgery, Department of Surgery, University of Ulsan College of Medicine and Asan Medical Center, 388-1 Pungnap2-dong, Songpa-gu, Seoul 138-736, Korea.
    • ,
  • S.-J. Park

      Affiliations

    • Department of Surgery, University of Ulsan College of Medicine and Ulsan University Hospital, Ulsan, Korea
    • ,
  • H.-K. Kim

      Affiliations

    • Division of Vascular Surgery, Department of Surgery, University of Ulsan College of Medicine and Asan Medical center, Seoul, Korea
    • ,
  • H.-K. Yoon

      Affiliations

    • Division of Interventional Radiology, Department of Radiology, University of Ulsan College of Medicine and Asan Medical center, Seoul, Korea
    • ,
  • G.-E. Kim

      Affiliations

    • Division of Vascular Surgery, Department of Surgery, University of Ulsan College of Medicine and Asan Medical center, Seoul, Korea
    • ,
  • B. Yu

      Affiliations

    • Division of Rheumatology, Department of Internal Medicine, University of Ulsan College of Medicine and Asan Medical Center, Seoul, Korea

Accepted 24 August 2007. published online 29 October 2007.

Article Outline

Objective

We report our surgical treatment results of abdominal aortic aneurysm (AAA) in Behçet's disease patient.

Materials and methods

Between September 1998 and June 2006, the authors have performed 21 procedures for AAA in 12 patients with Behçet's disease. Male to female sex ratio was 3:1 and mean age was 34 years old. Behçet's disease was diagnosed clinically using criteria of International Study Group for Behçet's Disease (1990). Retrospective analysis was made.

Results

There were six infrarenal, five suprarenal, and one double (suprarenal and infrarenal)AAA. Six graft interposition, six patch closure, and one stent-graft insertion were performed (one graft interposition and one patch closure were simultaneously performed for double AAA). Eight recurrent aneurysms were noted in six (50%) patients. Four stent-graft insertion, two patch closures, one graft interposition and one explothoracotomy only were performed for recurrent aneurysms. Overall recurrence rate of 21 procedures was 38.1%; 14.3% for graft interposition, 62.5% for patch closure, and 40% for stent-graft insertion.

Conclusion

Though the resection and graft interposition is technically difficult in many occasions, it should be considered as the procedure of choice for abdominal aortic aneurysm in Behçet's disease. Endovascular interventions may be one of the treatment modality but the result needs further long-term follow-up.

Keywords: Behçet's disease, Vasculitis, Abdominal aorta, Aneurysm, Endovascular surgery

 

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Introduction 

Because of high rupture rate, aneurysm of Behçet's disease should be repaired. However, recurrent aneurysm is frequently seen following surgical treatment. Many attempts have been tried to prevent recurrence including stent-graft treatment, but the recurrence or puncture site aneurysm can be problematic.1, 2, 3, 4, 5 There is still no definite medical treatment or widely accepted way of assessing disease activity.6, 7, 8

We have treated twelve Behçet's disease patients with abdominal aortic aneurysm by various treatment modalities and herein, report our surgical treatment results.

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Materials and Methods 

Between September 1998 and June 2006, the authors have treated twelve patients with Behçet's disease, who had abdominal aortic aneurysm (AAA). Male to female sex ratio was 3:1 and median age was 33.5 years old. The diagnosis of Behçet's disease was made clinically using criteria of International Study Group for Behçet's Disease (1990). Median follow-up period was 45.5 (4–98) months.

Abdominal aortic aneurysm was diagnosed on abdominopelvic CT (computerized tomographic) scan. All patients were followed and monitored by one of the authors (Bin Yu) regularly with one to three months interval postoperatively. Parameters used for the disease activity were relapsing symptoms, CRP(C reactive protein) and ESR (Erythrocyte sedimentation rate). As for imaging study, we used aortic dissection CT scan regularly for 6 months interval following surgery. Retrospective analyses of the clinical manifestations, treatment modalities, and outcome on these patients are reported.

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Results 

The symptoms and signs of the patients that might be clue in diagnosis of Behçet's disease were recurrent oral ulcer (100%), skin lesion (83.3%), recurrent genital ulcer (50%), and positive Pathergy test (50%), and eye lesion (33.3%), history of arterial surgery (25%), deep vein thrombosis (25%), and arthritis (25%). The elevation of erythrocyte sedimentation rate (ESR) or C-reactive protein (CRP) was observed in all but one patient (91.7%). Leukocytosis was noted in 8(66.7%). Among the 12 patients, two patients (16.7%) developed abdominal aortic aneurysm while on immunosuppressive medication for Behçet's disease and the other ten patients came without diagnosis of Behçet's disease. As for previous arterial lesions, there were three femoral artery aneurysms which had been treated by resection and interposition graft using prosthetic graft (Table 1).

Table 1. Patients' profile at the first presentation
Patient N=12SexAgeOral ulcerSkin lesionGenital ulcerPathergy testEye lesionDVTArterial diseaseArthritisPrior immuno suppressant
1M38YY + Iliac V
2M30YYY+ Iliac V
3M50YY −YIliac VFA
4F16YYY−
5M44YY −Y FA Y
6M35YY + Y
7M34YY −Y
8M38Y Y+
9F26YYY–
10M31Y +Y Y
11F33YYY−
12M34YYY+ FAYY

M:F=3:134*100%83.3%50%50%33.3%25%25%25%16.7%

DVT: Deep vein thrombosis; Y: Positive history; *: Mean age; V: Vein; FA: Previous femoral artery aneurysm.

All aneurysms were saccular in shape with median 40mm of maximal diameter and median 15mm of neck diameter on preoperative CT scan (Fig. 1). Infrarenal AAA was noted in 6 patients (50%) and suprarenal AAA was noted in 5 patients (41.7%). One patient (8.3%) had suprarenal and infrarenal abdominal aortic aneurysms (Fig. 2). Contained rupture was the most common manifestation of the aneurysm and noted in nine out of 12 patients (75%), followed by free rupture in 2 (16.7%), and occlusion of iliac artery in one patient (8.3%).

  • View full-size image.
  • Fig. 1 

    Typical feature of AAA (white circles) in Behçet's disease - saccular in shape with relatively short neck portion. Three dimensional reconstruction (A), sagital section (B), and axial section (C) on CT scan.

Because of urgency, all patients except one who had AAA and iliac artery occlusion were treated on emergency base. As for infrarenal AAA, four resection and aorto-iliac graft interposition, one stent graft, and one patch closure were performed. As for suprarenal AAA, four patch closure and one resection and graft interposition with visceral artery reimplantation were performed. In a patient with double AAA, patch closure for suprarenal AAA and resection and aortoiliac graft interposition for infrarenal AAA were performed simultaneously.

We have performed prosthetic wrapping on the proximal aortic anastomotic site in every cases of graft interposition to prevent anastomotic aneurysm. Omental wrapping was performed in recent two patients, who had infrarenal AAA and double aneurysms.

Systemic immunosuppressive medication including steroid, colchicines, azathioprine, or cyclophosphamide was given to all patients, postoperatively. Dosage and combination of the medicine were individually tailored.

There was no operative mortality. Mechanical ileus during immediate postoperative period requiring operative treatment occurred in two patients (16.7%). Two patients died during the follow-up period. Ruptured recurrent aneurysm was the cause of death in one patient four months after patch closure for suprarenal AAA and sepsis following necrotizing fasciatis was the cause of death in other patient at 21 months after graft interposition for infrarenal AAA. Recurrent aneurysm was noted in six (50%) patients. As for location of aneurysm, there were two recurrences out of seven (28.6%) infrarenal AAA and four recurrences out of six (66.7%) suprarenal AAA. Regarding the procedures, there was one recurrence out of six interposition graft (16.7%), four out of six patch closures (66.7%), and one stent graft insertion (100%).

There were 8 recurrences in 6 patients. Each patient had one recurrence except one who had multiple recurrences (three times). Median recurrence interval was 19(4–98) months.

Treatment for the eight recurrent lesions were four stent graft insertion (Fig. 3, Fig. 4, Fig. 5), two patch closure, one graft interposition and visceral artery reimplantation, and one explothoracotomy only. One patient who had three recurrences had been treated with redo patch closure for the first recurrence, stent graft treatment for the second recurrence, and interposition graft with visceral artery reimplantation for the third recurrence (Table 2).

Table 2. Characteristics of lesion, treatment and result
PatientLocation of AAASizeSymptomTreatment (graft size: mm & type)Compli-cationImmuno-suppresantRecurr-enceFollow Up durationResult
Maximal diameter (mm)Neck diameter (mm)
1Infra4015C. RuptureInterposition(PTFE)IleusPd+Col 98MAlive
16×8, bifurcated
2Infra5020C. RuptureInterposition(PTFE) Pd+Col 21MDeath
16×8, bifurcated
3Infra6020OcclusionInterposition(PTFE) Pd+Col+Aza 66MAlive
14, straight
4Infra3510C. RuptureInterposition(PTFE) Pd+Col 13MAlive
14×7, bifurcated
5Supra7010F. RupturePatch(PTFE) Pd+Cyc 48MAlive
6Supra2515C. RupturePatch(PTFE) Pd+Col 4MAlive
6 (Double)Infra3015 Interposition(PTFE)
18×9, bifurcated
7Supra5050C. RuptureInterposition(Dacron)* Pd+Col+Aza18M92MAlive
16, straight
7(R)Distal anast. RecurrentStent graft Pd+Col+Aza
18, straight
8Infra3520C. RupturePatch(PTFE) Pd+Col11M31MAlive
8(R)Patch site RecurrentStent graft Pd+Col+Cyc
18, straight
9Supra5010C. RupturePatch(PTFE) Pd+Col+Aza8M73MAlive
9(R)Patch site RecurrentPatch(PTFE) Pd+Col+Aza17M
9(R)Patch site RecurrentStent graft Pd+Col+Cyc32M
20, straight
9(R)Stent graft distal RecurrentInterposition(PTFE)* Pd+Col+Cyc
22, straight
10Supra5015RupturePatch(PTFE)IleusCol+Aza18M43MAlive
10(R)Patch site RecurrentPatch(PTFE) Pd+Aza
11Supra2510C. RupturePatch (Dacron) Pd+Cyc5M4MDeath
11(R)Patch site RecurrentExplothoracotomyOp death
12Infra3520C. RuptureStent graft Pd+Col+Aza19M52MAlive
18, straight
12(R)Stent graft proximal RecurrentStent graft Pd+Col+Aza
22, straight
Total 21 Median 40mmMedian 15mm Median 45.4M

Supra: Suprarenal; Infra: Infrarenal; (R): Recurrent; *: Graft interposition with visceral artery reimplantation; M: Months; Anast: anastomosis; C.: Contatined; F.: Free; Pd: Prednisolone; Aza: Azathioprine; Col: Colchicines; Cyc: Cyclophosphamide.

Overall recurrence rate by the procedure (21 procedures) was 38.1%. Recurrence rate of the resection with interposition graft, patch closure, and stent-graft insertion were 14.3% (1/7), 62.5% (5/8) and 40% (2/5), respectively (Table 3).

Table 3. Recurrence rate according to the procedure or location of the aneurysm
Graft interpositionPatch closureStent graftExplothoracotomyRecurrence rate (%)
Suprarenal AAA1/13/5 4/6 (66.7)
Infrarenal AAA0/51/11/1 2/7 (28.6)
Recurrent aneurysm0/11/21/40/12/8 (25)
Recurrence rate (%)1/7(14.3)5/8(62.5)2/5(40)0/18/21(38.1)

There were no significant statistical differences in recurrence rate between sex, age, and clinical manifestation, location of the aneurysm, ESR, CRP and postoperative immunosuppressive medication. However, those with interposition showed less recurrence than patch (p=0.0389) with statistical significance and showed the best result among the three procedures (Fig. 6).

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Discussion 

Behçet's disease is a chronic recurrent inflammatory disorder affecting many organs. Patients are usually 20–30 years old at the time of onset of the disease.9

The clinical course of Behçet's disease is characterized by remissions and exacerbations that generally abate in intensity with the passage of time. There is no universally accepted diagnostic test or accepted way of assessing disease activity.6, 7 Thus, the diagnosis of Behçet's disease has relied on identification of several of its more typical clinical features. At present, the diagnosis is made on the basis of the criteria proposed by the International Study Group for Behçet's Disease in 1990.10

The spectrum of vascular disease is broad and unique in Behçet's disease. It is one of the few vasculitis that can involve large vessels, both arteries and veins. The frequency of vascular involvement in Behçet's disease is estimated 2 to 46% and it is four to five times more common in men in endemic region.1, 11, 12, 13, 14, 15 Vascular lesions seen in Behçet's disease are venous thrombosis, arterial thrombosis, and arterial aneurysm. Inflammation of the aorta, pulmonary and peripheral arteries is one of the most serious complications of Behçet's disease and is responsible for the majority of deaths.9, 16 Arterial lesions are less common than venous disease and its prevalence is about 1.5 to 3% worldwide. Aneurysm is reported to be more common than occlusion and aneurysm formation is seen in almost all named arteries but the abdominal aorta is the most common site. Rupture is the most common complication of aneurysm and the most common cause of vascular related death.2, 9, 13, 17 Multiple aneurysms are relatively common.18, 19

Exact incidence of arterial lesion or aneurysm in Behçet's disease in Korea is not known. In our small series, aneurysm was more common than occlusion, and most commonly seen was the abdominal aortic aneurysm. Aneurysm in Behçet's disease is different from that of degenerative type in many aspects. The age incidence is younger, suprarenal type is more commonly seen, shape of aneurysm is usually saccular, multiple aneurysm is more frequently seen, and symptom is more urgent.

Of all the vascular lesions in Behçet's disease, aneurysm is the most complicated and challenging pathology for the vascular surgeon because of the technical difficulties and frequent recurrence.20, 21, 22, 23

Since the first report of an abdominal aortic aneurysm due to Behçet's disease, by Mishima24 in 1961, many attempts have been made to surgically manage these serious arterial lesions. However, these were a case report or experience with small numbers of patients.

Some authors insisted that the establishment of remission by use of glucocorticoid before the surgical intervention can decreases the incidence of postoperative anastomotic complications.25 Because of urgency, we performed surgery on emergency base so we could not use preoperative steroid. Two patients in our series developed abdominal aortic aneurysm while on immunosuppressive medication for Behçet's disease. It has been reported that rupture is not correlated to the size of aneurysm in Behçet's disease26 and aneurysm should be repaired at the time of detection because of high rupture risk. This is true in our series that all aneurysms were saccular in shape with median 40mm of maximal diameter relatively short neck and contained rupture was the most common indication for treatment.

Some authors have suggested performing a bypass with anastomoses on “intact” arteries, far away from the aneurysms, or performing an extraanatomic bypass.27, 28 Others suggested simple aneurysmectomy or closure of the arterial defect by direct suture or patching.3 Actually, we performed patch closure mainly in suprarenal AAA because of urgency and technical difficulties while we performed resection and interposition graft mainly in infrarenal AAA. So it's not easy to compare these two procedures but patch closure showed statistically significant higher recurrence rate than resection and interposition graft in our series. Performing a bypass with anastomoses on “intact” arteries seems to be as the procedure of choice for abdominal aortic aneurysm in Behçet's disease. Arterial protection by prosthetic wrapping has been reported.3 We have also performed prosthetic wrapping on the proximal aortic anastomotic site which seemed to be effective in preventing recurrence. But considering iliac anastomotic site which was not wrapped, we could not evaluate the exact role of this prosthetic wrapping. Recently, we have performed omental wrapping over the graft or patch after resection of the aneurysm as Tüzün reported.2 Above is not an evidence based procedure but it may be helpful in decreasing the dead space and alleviating the residual inflammation following resection of the aneurysm.

Recently, stent graft treatment has been reported as one of the treatment modality for an aneurysm to avoid a recurrent aneurysm at the anastomosis site which was commonly seen following surgical treatment.26, 29, 30

We have performed five stent graft treatments (one for original lesion and four for recurrent aneurysms). Stent grafts used were custom-made S & G straight stent grafts with proximal and distal bare stents for fixation. All stent grafts were deployed through 14 French sheath which was introduced via one femoral puncture.Femoral puncture sites were managed by manual compression. There was no initial or late endoleak and there was no early or late puncture site problem. However, recurrent aneurysms occurred at the site just proximal or distal to stent graft in two patients where the aorta was thought to be normal previously, were noted (40% of recurrence rate). No femoral pseudoaneurysm developed at the puncture site during the follow-up period. The main advantages of endovascular intervention are low mortality rates (0.6%–3.5%) and high technical immediate success rates.31 However, experience with the endovascular treatment is still limited in Behçet's disease patient. Furthermore, one of the most important aspects of Behçet's disease is aneurysmal arteriopathy and its tendency to develop recurrent false aneurysm at angiographic puncture site.1, 2, 3, 4, 5 Though there was no puncture site aneurysm in our series, it should be performed in selected cases with caution.32

Some authors supported a surgical approach with simple ligature of the artery to avoid complications of reconstructive surgery in peripheral arterial lesions.2 It seemed to be true especially for aneurysm at superficial femoral artery. In our series, there were three femoral artery aneurysms that had been treated by resection and prosthetic graft interposition but all subsequently occluded without symptom of critical limb ischemia.

Immunosuppressant and anti-inflammatory medication should be conducted for suppression of the vasculitis.7 However, the role of immunosuppressant and anti-inflammatory medication for prevention of the recurrent arterial lesion following intervention in Behçet's disease also needs to be further evaluated.

In conclusion, resection and interposition graft showed much better result than patch closure for AAA due to Behçet's disease with statistical significance in our series. Though the resection and graft interposition is technically difficult in many occasions, it should be considered as the procedure of choice for abdominal aortic aneurysm in Behçet's disease. Endovascular interventions may be one of the treatment modality but the result needs further long-term follow-up. Because of high recurrence rate and lack of specific tool for early diagnosis of recurrence, at present, close follow-up with regular interval is the only way to be recommended for detection and treatment of recurrent AAA in patients with Behçet's disease.

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References 

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PII: S1078-5884(07)00556-4

doi:10.1016/j.ejvs.2007.08.013

European Journal of Vascular & Endovascular Surgery
Volume 35, Issue 2 , Pages 173-180, February 2008

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