Annals of Vascular Surgery
Volume 20, Issue 4 , Pages 464-471, July 2006

Endovascular Treatment of Complicated Aortic Aneurysms in Patients with Underlying Arteriopathies

Division of Vascular Surgery, Department of Surgery, Mount Sinai School of Medicine, New York, NY, USA

Article Outline

Patients with arteriopathies including giant cell arteritis, Marfan syndrome, and Takayasu's disease are at risk for aneurysmal degeneration of the aorta. Aortic repair has been recommended for these patients to prevent rupture. The purpose of this study was to examine outcomes following endovascular stent graft (EVSG) repair of aortic aneurysms in this patient population. Over an 8-year period, 11 patients (six men, five women) with arteriopathies underwent endovascular aortic repair. The mean age was 50 (range 15–81). Diseases included Marfan syndrome (n = 6), Takayasu's disease (n = 3), and giant cell arteritis (n = 2). Success of EVSG repair was evaluated per the reporting standards of the Society for Vascular Surgery/American Association for Vascular Surgery. Follow-up was a mean of 28.9 months (range 3–68). Six patients underwent EVSG repair of the thoracic aorta, four underwent EVSG repair of the abdominal aorta, and one underwent a staged repair of the thoracic and subsequently the abdominal aorta. Six true aneurysms and six pseudoaneurysms were repaired. Eight patients had previous aortic surgery, including four with multiple aortic operations. For the 12 aneurysms treated, technical success was achieved in 11 (91.7%). One technical failure occurred due to a small iliac access vessel, requiring an eventual iliac conduit for insertion. Early complications (<30 days) occurred in three patients. Type I or III endoleak developed following two repairs (16.7%). Aneurysm expansion occurred following one repair (8.3%). No aneurysm-related deaths occurred during follow-up. EVSG repair of aortic aneurysms is feasible and can be safely performed in patients with arteriopathies. Long-term durability in this younger group of patients who carry an ongoing risk of arterial degeneration remains to be determined.

 

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INTRODUCTION 

Aortic aneurysms are most commonly associated with atherosclerotic disease; however, they can result from a number of different etiologies. In particular, patients with underlying arteriopathies, including Marfan syndrome, Takayasu's disease, and giant cell arteritis, are at risk for aneurysmal degeneration of the aorta. Aneurysm formation in Marfan patients occurs as a result of a mutation in the fibrillin gene leading to weakening of the elastic layers of connective tissue.1, 2 For patients with giant cell arteritis and Takayasu's disease, aneurysmal degeneration is thought to be a late complication resulting primarily from destruction of elastic tissue in the arterial wall.3, 4 As with patients who develop aortic aneurysms from atherosclerotic disease, repair has been recommended for patients with aneurysms resulting from arteriopathies to prevent rupture.

Endovascular stent graft (EVSG) repair of aortic aneurysms has been shown to be a safe and efficacious alternative to open repair.5, 6, 7 Similarly, EVSG repair of para-anastomotic aneurysms has been shown to be a safe and efficacious alternative to open repair.8, 9 Patients with arteriopathies who develop aneurysms typically require multiple aortic operations to treat the aneurysms at various levels. For such patients, EVSG repair offers a less invasive and possibly safer alternative to open repair for the treatment of both primary and para-anastomotic aneurysms. We reviewed our experience with EVSG repair of aortic aneurysms in patients with arteriopathies to evaluate outcomes in this group.

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METHODS 

A retrospective review of all patients undergoing EVSG repair at our institution between January 1997 and July 2005 was performed. Of 1,073 patients who underwent EVSG repair, 11 (six men, five women) with arteriopathies were identified. Diseases included Marfan syndrome (n = 6), Takayasu's disease (n = 3), and giant cell arteritis (n = 2). All patients were treated in accordance with the institutional review board of Mount Sinai Medical Center.

Prospectively gathered data, including demographics, previous aortic procedures, aneurysm size, medical comorbidities, success of aneurysm repair, complications, secondary interventions, and survival, were investigated. Anatomic characteristics of aneurysms with regard to maximal diameter as well as suitability for endovascular repair were based on preoperative contrast-enhanced computed tomography (CT) and, in select cases, arteriography. All procedures were performed in the operating room with portable C-arm fluoroscopy. Follow-up included an office visit with the operating surgeon in addition to anteroposterior and lateral abdominal radiographs and contrast-enhanced CT angiograms (CTAs) at 1, 6, and 12 months after hospital discharge and yearly thereafter.

Success, both technical and clinical, was identified according to the Society for Vascular Surgery/American Association for Vascular Surgery (SVS/AAVS) reporting standards. Technical success required successful access to the arterial system using a remote site, successful deployment of the endoluminal graft with secure proximal and distal fixation, and no evidence of type I or type III endoleak. Type II endoleaks were not considered technical failures. Clinical success required successful deployment without death as a result of aneurysm-related treatment, freedom from type I or type III endoleaks, no aneurysm expansion (>5 mm), rupture, graft failure, graft migration, or conversion to open repair. Aneurysm-related death included any death resulting from aneurysm rupture, a primary or secondary endovascular procedure, or surgical conversion.

This group of patients was treated with five different endovascular devices. Talent thoracic devices (Medtronic, Santa Rosa, CA) were implanted in seven patients. A Talent bifurcated device was implanted in one patient, and a Talent aorto-uni-iliac device was implanted in one patient. Bifurcated devices (Gore and Associates, Flagstaff, AZ) were implanted in two patients. An AneuRx tube device (Medtronic) was implanted in one patient.

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RESULTS 

Eleven patients with a mean age of 50 years (range 15–81) were treated with EVSG repair during the 8-year period that was reviewed. Six patients underwent EVSG repair of the thoracic aorta, and four underwent EVSG repair of the abdominal aorta. One patient underwent a staged repair of the thoracic aorta followed 8 months later by repair of the abdominal aorta. Six true aneurysms and six pseudoaneurysms were repaired (Fig. 1). The six pseudoaneurysms included five patients with paraanastomotic aneurysms and one patient with a saccular aneurysm. Eight of the 11 patients (72.7%) had undergone previous aortic surgery, including four who had had multiple previous aortic operations. Of the seven patients with thoracic aortic aneurysms, only one had arch involvement, which necessitated coverage of the left subclavian artery (Table I, Table II).

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  • Fig. 1. 

    Arteriogram demonstrating a pseudoaneurysm at the distal suture line in a patient with giant cell arteritis who had undergone three previous thoracic aortic repairs. B CTA demonstrating infrarenal abdominal aortic aneurysm in the same patient. C Three-dimensional reconstruction of CTA demonstrating aneurysm exclusion following successful thoracic and abdominal aortic repair.

Table I. Patient demographics
PatientAge (years)/sexArteriopathy diagnosisAdditional comorbiditiesPrevious aortic surgery
178 FGiant cellCAD, HTN, breast cancerNone
262 FGiant cellCAD, HTN, COPDOpen TAA repair (×3)
344 MMarfanCAD, HTN, COPDBentall, TAA repair
481 MMarfanHTN, CVABentall
548 MMarfanHTN, paraplegiaBentall, open AAA repair
640 MMarfanCOPDOpen TAA repair
751 MMarfanHTNBentall
825 MMarfan-Open AAA repair
915 FTakayasu'sHTNNone
1056 FTakayasu'sHTNNone
1145 FTakayasu's-Open TAA repair (×2)

CAD, coronary artery disease; COPD, chronic obstructive pulmonary disease; CVA, cerebrovascular accident; HTN, hypertension; AAA, abdominal aortic aneurysm; TAA, thoracic aortic aneurysm.

Table II. Aneurysm and device characteristics
Device diameters (mm)
PatientAneurysmInitial sac size (mm)Endovascular deviceNumber of devices used (thoracic)ProximalDistal
1TAA (true)57Talent thoracic23430
2TAA (pseudo)86Talent thoracic34438
AAA (true)55Gore bifurcatedn/a2612
3TAA (pseudo)95Talent thoracic34640
4AAA (true)49Gore bifurcatedn/a2620
5AAA (pseudo)70Talent aorto-uni-iliacn/a3612
6TAA (pseudo)48Talent thoracic13838
7AAA (true)55Talent bifurcatedn/a3220
8RCIAA (true)48AneuRx tuben/a1515
9TAA (true)44Talent thoracic22420
10TAA (pseudo)72Talent thoracic12416
11TAA (pseudo)55Talent thoracic12420

AAA, abdominal aortic aneurysm; TAA, thoracic aortic aneurysm; RCIAA, right common iliac artery aneurysm; n/a, not applicable.

Three patients underwent procedures in preparation for their EVSG repairs: two underwent internal artery embolization and one, who had developed a visceral patch pseudoaneurysm from a previous open thoracoabdominal repair, underwent bypasses to the celiac and superior mesenteric arteries from the proximal graft as well as an iliorenal bypass. Initial technical success was achieved in 11 of 12 cases (91.7%). One patient whose initial operation was aborted due to difficulty advancing the stent graft went on to a successful repair utilizing an iliac condult. Of note, a total of three patients required iliac conduits to allow for device deployment. Of these three patients, two had had multiple thoracic aortic procedures requiring cardiopulmonary bypass and femoral artery cannulization.

Early complications (<30 days) occurred following three procedures (25%). One patient had a postoperative myocardial infarction and one patient developed a wound infection requiring incision and drainage. Patient 5, a Marfan patient, developed a left femoral artery pseudoaneurysm following EVSG repair of an aortic pseudoaneurysm with an aorto-uni-iliac device. This required revision of the femoral-femoral bypass anastomosis and an axillofemoral bypass secondary to iliac artery dissection at the time of revision. Ultimately, he required excision of the femoral-femoral bypass due to infection. Arterial flow to his left lower extremity was maintained via a bypass from the axillofemoral graft to the left superficial femoral artery. There were no perioperative mortalities or immediate conversions to open procedures. The mean length of stay was 2.75 days (range 1–10).

Mean follow-up was 28.9 months (range 3–68), during which time there were no aneurysm-related deaths. Late complications occurred following three operations (25%), which required additional aortic procedures. Patient 3 developed a type III endoleak, which was successfully treated with placement of an additional Talent thoracic device 43 months after his initial EVSG repair. However, he ultimately had a contained rupture of the intercostal patch from his original open thoracoabdominal repair, necessitating a second open repair 9 months after the repair of his type III endoleak. Patient 10 developed a type I endoleak, which was detected 3 months postoperatively and successfully treated with percutaneous angioplasty (Fig. 2). Patient 8, who had an AneuRx tube placed for an aortoiliac aneurysm, developed claudication of this limb. On angiography, a stenosis was noted within the stent graft at the anastomotic line from his original open abdominal aortic aneurysm repair. This was successfully managed with balloon dilatation of the stent graft.

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  • Fig. 2. 

    CTA in a patient with Takayasu's disease demonstrating a saccular aneurysm arising from a densely calcified aorta. B Postoperative CTA with type I endoleak. C Percutaneous treatment of the type I endoleak with evidence of a calcified aorta as shown by the constrained midportion of the angioplasty balloon.

Three patients underwent open aortic repair in the follow-up period distinct from their endovascular repairs. The following three patients' endovascular repairs remained intact; however, they required open aortic operations at separate anatomic locations, demonstrating the systemic effects of these arteriopathies. Patient 6, who initially underwent EVSG repair of an intercostal patch pseudoaneurysm following open repair of a descending thoracic aortic repair, developed a pseudoaneurysm at the distal anastomosis of his open thoracic aortic repair as well as aneurysmal degeneration of the visceral segment. This required open thoracoabdominal repair 28 months following his EVSG repair. Patient 11, who had undergone two previous thoracic aortic repairs, underwent endovascular repair of a thoracic pseudoaneurysm of the distal anastomosis. She subsequently developed a pseudoaneurysm of the proximal anastomosis requiring open repair. Patient 8 underwent endovascular treatment of a common iliac artery aneurysm. He previously had undergone an open repair of his infrarenal abdominal aorta. Due to progression of his disease, he required not only thoracoabdominal aortic repair but arch repair as well.

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DISCUSSION 

Arteriopathies, including giant cell arteritis, Marfan syndrome, and Takayasu's disease, are associated with aneurysmal degeneration of the aorta. In patients with Marfan syndrome, aneurysm formation is the result of a mutation in the fibrillin gene leading to weakening of the elastic layers of connective tissue.1, 2 In both giant cell arteritis and Takayasu's disease, aneurysmal degeneration is thought to be a late complication resulting from destruction of the arterial wall and its elastic tissue.3, 4 For patients with these arteriopathies, the presence of aortic aneurysms increases their overall morbidity and may reduce long-term survival.10, 11

Patients with Marfan syndrome are at risk of a range of cardiovascular complications, including aortic valve regurgitation, aortic dissection, and aortic aneurysm. Long-term surveillance of the entire aorta and repair, when indicated, has become standard practice because it has been shown that survival is prolonged in Marfan patients who undergo prophylactic aortic operations.12, 13, 14

Given the benefits of prophylactic aortic surgery, particularly of ascending aortic aneurysms and the aortic valve, there is a growing number of patients who may require secondary aortic operations as a result of anastomotic failures, new dissections, or new aneurysms. Additionally, it has been shown that Marfan patients have a disproportionately high anastomotic failure rate.15 With the increased risk of repeat aortic surgery and the benefits of endovascular therapy, EVSG repair has emerged as a feasible alternative to open repair for these patients. Endovascular stent grafts have been successfully used to treat persistent distal aortic dissection after previous root replacement, as reported by Ince et al.16 In their series of six patients, two went on to require elective open conversion. Other than this series, only a few other case reports have reported the use of stent grafts to treat dissection17 and a thoracoabdominal aneurysm18 in Marfan patients. In our series, six patients with Marfan syndrome, all of whom had undergone previous aortic operations, underwent technically successful endovascular aortic repair.

The development of aortic aneurysms occurs less frequently in Takayasu's disease than in Marfan syndrome. Takayasu's disease, which affects primarily young Asian, African, and South American women, is more commonly characterized by an early stage which clinically presents as a systemic inflammatory illness, followed by a late occlusive stage which presents as arterial insufficiency. However, 2–45% of Takayasu's disease patients develop aortic aneurysms.19, 20, 21, 22 These aneurysms occur most commonly in the thoracic and thoracoabdominal aortae.23 Although older patients with long-standing disease are at greater risk for the development of aneurysms, aneurysms may be found in younger patients as well.20 This was evident in our series, where a 15-year-old female was treated for a descending thoracic aortic aneurysm.

In addition to destruction of the arterial wall and its elastic tissue, hypertension and poststenotic dilatation are thought to play a role in the development of aneurysms in patients with Takayasu's disease.24 Furthermore, the rate of growth of aneurysms in these patients is thought to be related to persistent hypertension.22 Of note, in our series, two of the three patients with Takayasu's disease had hypertension, for which they were receiving treatment. However, the rate of growth of aneurysms in these patients is slower than in those with atherosclerotic aneurysms due to the aortic scarring associated with the disease.22 Regardless, these aneurysms are thought to carry the same potential for rupture and embolization as aneurysms from other causes.23

Patients with Takayasu's disease who develop aneurysms may be at higher operative risk for conventional repair for a number of reasons. The vast majority of these patients are treated with long-term steroid therapy and carry the risks that go along with this. At the same time, it has been advocated that control of inflammation is mandatory before surgical intervention to control the inflammatory reaction from surgery and to prevent anastomotic insufficiency.4, 25 Despite this approach, anastomotic aneurysms occur at a higher incidence in Takayasu's disease than in atherosclerosis and are more likely to occur in patients who undergo surgery for aneurysmal lesions rather than occlusive lesions.26 For these reasons, EVSG repair may be beneficial in order to avoid anastomoses in the setting of inflamed arteries and to minimize surgical trauma. EVSG repair has been used for both aneurysmal disease23, 27, 28 and stenotic disease on a limited basis. Our two Takayasu's disease patients with true thoracic aortic aneurysms were successfully treated and have been followed for 28 and 48 months with markedly smaller aneurysms compared to preoperatively. The Takayasu's patient with a thoracic aortic pseudoaneurysm who underwent EVSG demonstrated the high-risk nature of such patients when she developed an aortotracheal fistula from the proximal anatomosis of her open thoracic repair, necessitating tertiary open thoracic aortic repair.

Giant cell arteritis, which affects large and medium-sized arteries in persons 50 years or older predominantly of northern European ancenstry, most commonly presents with symptoms related to inflammation of the distal branches of the extracranial carotid arteries, including headache, jaw claudication, and blindness. Additionally, aortic aneurysms resulting from previous damage are reported to occur in 18% of patients with giant cell arteritis.29 Furthermore, patients with giant cell arteritis are 17.3 times more likely to develop a thoracic aortic aneurysm and 2.4 times more likely to develop an abdominal aortic aneurysm than the general population.30 The time from diagnosis of giant cell arteritis to the detection of thoracic aortic aneurysms or abdominal aortic aneurysms in a large-population study was 10.9 and 6.3 years, respectively.29 Although the development of aneurysms typically occurs years after the initial diagnosis of giant cell arteritis, it is important to note that aneurysm development may be the initial presenting sign of the disease.

Patients with giant cell arteritis, unlike patients with Takayasu's disease, are often elderly and have multiple comorbidities, increasing the risk of open aortic repair. As for patients with atherosclerotic aortic aneurysms, EVSG repair offers the same benefits from its less invasive nature for these patients. Additionally, as with Takayasu's disease, patients with giant cell arteritis may be at risk of anastomotic failures given the underlying pathology of the arteries, perhaps making EVSG repair a better option. Only two other reports have described EVSG repair in patients with giant cell arteritis, which were performed for an aorto-esophageal fistula associated with a thoracic aneurysm31 and a thoracic aneurysm with an intramural hematoma.32

Special consideration should be given to patients with all three of these arteriopathies before undertaking EVSG repair. Prior to repair, it should be confirmed that there is no other etiology to account for aneurysm development and, in particular, that the aneurysm is not mycotic in nature. This may be of particular relevance for patients who have been maintained on chronic steroid therapy to treat their arteriopathies. Simultaneously, patients with inflammatory arteriopathies should have medical optimization of their inflammation prior to repair. Additionally, these patients should be investigated for the presence of concomitant aneurysms throughout their arterial systems, given the reported occurrence of multiple aneurysms.33

In our series, the rates of both early and late complications appear to be higher than those following endovascular repair of atherosclerotic aneurysms. Given this patient population, these figures result from a variety of different factors including the underlying pathologies, the high proportion of patients undergoing reoperations, and finally, the small number of patients. All of these arteriopathies carry an ongoing risk of arterial degeneration, as demonstrated in a series of open repairs and by the significant proportion of patients in our study who required additional aortic operations. This appears to be particularly true of patients with Marfan syndrome, who made up the majority of patients requiring reintervention, both directly related to their endovascular repairs and for aneurysms involving their native aortas. Although the use of stent grafts to treat these patients obviates the need for aortic anastomoses, they are still at risk for landing zone failures, endoleaks, and subsequent aneurysm growth. It is impossible to know which of these adverse events is more likely to occur, but it seems reasonable that EVSG repair may, at the very least, serve as a temporizing therapy. Furthermore, given the systemic nature of these diseases, EVSG repair may serve as definitive therapy and obviate the need for open repair for at least a portion of the aorta. Regardless, patients with any of these arteriopathies warrant lifelong surveillance of not only their endografts but their untreated aortas as well.

This series demonstrates that EVSG repair in patients with arteriopathies can be performed safely and efficaciously. Mid-term results are favorable; however, these patients are at continued risk for aneurysmal degeneration throughout their aortas, and many will ultimately require secondary aortic operations, both related and unrelated to their previous operations. With the advancing technology of EVSG repair and, in particular, the development of branched and fenestrated grafts the therapeutic options for these complex patients will continue to expand. At present, the long-term durability of endovascular aortic repair in this younger patient group remains to be determined, but it appears that select patients will benefit from the less invasive nature of EVSG repair.

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 This paper was presented at the Peripheral Vascular Surgery Society Winter Meeting, Park City, UT, January 26–29, 2006.

PII: S0890-5096(06)61463-5

doi:10.1007/s10016-006-9091-2

Annals of Vascular Surgery
Volume 20, Issue 4 , Pages 464-471, July 2006

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