Percutaneous Revascularization of Sole Arch Artery for Severe Cerebral Ischemia Resulting from Takayasu Arteritis

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1 Percutaneous Revascularization of Sole Arch Artery for Severe Cerebral Ischemia Resulting from Takayasu Arteritis Sanjay Tyagi, MD, Mohit D. Gupta, MD, Praveen Singh, MD, Devendra Shrimal, MD, and M.P. Girish, MD PURPOSE: Takayasu arteritis (TA) is a rare form of inflammatory arteriopathy affecting the aorta and its major branches. Obstructive lesions of all arch vessels may lead to disabling neurologic symptoms. There is limited experience with endovascular revascularization in this situation. The present report describes immediate and follow-up results with stent-supported angioplasty of severely stenosed single patent arch arteries. MATERIALS AND METHODS: Angioplasty and stent placement were performed in 10 consecutive patients with TA (age, years; mean, 28.3 y 4.1) with cerebrovascular symptoms caused by severe stenotic lesions of supraaortic (ie, carotid, vertebral, and brachiocephalic) arteries. RESULTS: Angioplasty was attempted in 12 stenotic lesions: carotid (n 8), vertebral (n 2), and brachiocephalic (n 2). Eight lesions were discrete and four were diffuse. Balloon angioplasty and stent implantation were performed successfully in all patients. The luminal diameter increased from 0.84 mm 0.6 to 5.6 mm 0.7 (P <.001), and the diameter of stenosis was reduced from 81.1% 6.8% to 1.7% 3.3% (P <.001). No immediate procedure-related complication or neurologic deficits occurred. The symptoms improved in all patients. On follow-up (mean, 25 months 7; range, 3 49 months), eight patients were asymptomatic and two patients with diffuse stenosis had recurrence of neurologic symptoms. These patients had in-stent restenosis of the carotid artery, which was successfully treated with cutting balloon angioplasty. CONCLUSION: Stent-supported angioplasty of a sole supraaortic artery in TA is safe and effective and provides good symptomatic relief in patients with multiple stenoocclusive lesions of arch arteries. J Vasc Interv Radiol 2008; 19: Abbreviation: TA Takayasu arteritis TAKAYASU arteritis (TA) is an uncommon chronic, progressive inflammatory disease affecting the aorta, its major branches, and sometimes the pulmonary arteries (1). Stenosis and occlusion of supraaortic arteries is common. The subclavian artery is From the Department of Cardiology, G. B. Pant Hospital and Maulana Azad Medical College, Suite 125, First Floor Academic Building, New Delhi , India. Received January 14, 2008; final revision received September 5, 2008; accepted September 14, Address correspondence to S.T.; drsanjaytyagi@vsnl.com None of the authors have identified a conflict of interest. SIR, 2008 DOI: /j.jvir most frequently involved, followed by proximal segments of the brachiocephalic, carotid, and vertebral arteries; and the disease may manifest as brain and retinal ischemia (2). As a result of the slow, progressive nature of the disease, collateral vessel formation is common. Therefore, symptoms of cerebral ischemia are most often associated with obstructive lesions of multiple arch vessels. It may manifest as fainting spells on standing, altered sensorium, seizures, vertigo (especially when looking upward), or visual disturbance (2 4). Medical management with glucocorticoids and immunosuppressive agents is ineffective in reducing stenosis in the chronic phase. Diffuse, proximal, and multifocal involvement of the arch vessels in this disease makes surgical revascularization difficult. Surgical repair of stenoocclusive lesions at the origin of arch vessels is much more complex and requires an intrathoracic approach, as opposed to endarterectomy of cervical carotid bifurcation stenosis in atherosclerosis. Lesions in aortoarteritis are distinctly characterized by more diffuse involvement and require higher balloon pressures to dilate, with more residual stenosis and a higher restenosis rate (5). Angioplasty has emerged as the initial mode of treatment for aorta, renal, and subclavian artery stenosis caused by aortoarteritis (3,4,6). However, the role of this technique in the treatment of carotid, brachiocephalic, and vertebral artery lesions is limited to few reports 1699

2 1700 Revascularization of Arch Artery for Ischemia in Takayasu Arteritis December 2008 JVIR (7 9). We attempted to achieve palliation of cerebral symptoms by angioplasty of a sole severely stenotic arch artery to improve cerebral blood flow while avoiding a totally occluded artery. We report our initial and follow-up results of angioplasty and stent placement in the proximal carotid, vertebral, and brachiocephalic arteries. MATERIALS AND METHODS We retrospectively analyzed all patients with TA who presented to our institution between January 2000 and June 2006 (N 14) with symptoms suggestive of severe cerebral ischemia. The diagnosis of TA was based on clinical, laboratory, and angiographic findings as established by the Aortitis Syndrome Research Committee of Japan (10). There were four male subjects and six female subjects whose ages ranged from 11 to 42 years (mean, 28.3 y 4.1). Three patients had recurrent syncope and seven had presyncope as a presenting symptom. One patient had associated seizures. Three of these patients also reported blurring of vision. Four patients were in the active phase of disease, as evidenced by an increased erythrocyte sedimentation rate. C-reactive protein was also increased in two of these patients. These four patients were administered glucocorticoids before their angioplasty procedures, but angioplasty was not delayed for more than 1 week in these patients as they had severe symptoms. A baseline angiographic study was performed to determine the extent of vascular disease. Patients underwent diagnostic aortic arch and abdominal aortography via a femoral approach under local anesthesia. Selective angiography of the stenosed artery was then performed to localize the site and extent of vessel involvement and distal circulation. Stenoses less than 2 cm in length were classified as discrete and those 2 cm or longer as diffuse. Angioplasty was performed in 10 patients for artery with severe stenosis ( 70%), and angioplasty was not attempted in vessels with total occlusion. Patients with subclavian artery angioplasty are not included in this series. Four patients with occlusion of all arch vessels were referred for surgical revascularization and are not included in this study. For the intervention, a 7-F, 90-cmlong Shuttle Flexor sheath (Cook, Bloomington, Indiana) was advanced over a 5-F cerebral diagnostic catheter into the proximal portion of the stenosed artery. Intravenous heparin (5,000 IU) was administered to maintain an activated clotting time of seconds during the procedure. The stenotic lesion was crossed with a floppy-tipped inch coronary guide wire, taking care not to enter the intracranial segment of the artery. The lesions were initially dilated with a monorail balloon 4 6 mm in diameter. The balloon was inflated for seconds with diluted contrast medium until the waist on the balloon disappeared or the rated burst pressure of the balloon was reached. Peripheral cutting balloons (4 6 mm diameter; Boston Scientific, Natick, Massachusetts) were used in four patients for initial dilation during the latter half of the study after these balloons became available. After balloon dilation, angiogram was obtained to assess the results. Balloon-expandable stents (5 7 mm in diameter; Express Vascular; Boston Scientific) were implanted in nine lesions, and self-expandable 7 9-mm nitinol stents (Smart Stent; Cordis, Miami Lakes, Florida) were implanted in three lesions. If the result was suboptimal after stent deployment, postdilation was performed with a high-pressure noncompliant balloon. No distal or proximal protection device was employed because these lesions are fibrotic, firm, and nonulcerated. All patients were given aspirin 150 mg/d and clopidogrel or ticlopidine daily for at least 3 days before the intervention, and this combination antiplatelet regimen was continued for 6 months. Thereafter, only aspirin 150 mg/d was given. Patients with evidence of disease activity as suggested by increased erythrocyte sedimentation rate were given oral steroids. Angioplasty was considered to be technically successful if the residual stenosis was less than 20% of the luminal diameter with at least a 50% increase compared with the pretreatment diameter. Patients were followed up at 1 month after treatment and then after 6 months, 12 months, and yearly. At each of these visits, independent neurologic examination was carried out by a neurologist. We recorded all procedure-related complications; transient ischemic attacks; nondisabling, disabling, and fatal strokes in the vertebrobasilar or carotid territory; myocardial infarctions; and deaths from any cause. The degree of residual stenosis immediately after endovascular treatment was measured on a posttreatment catheter angiogram. Patients were further followed up with Doppler ultrasound (US) examination at 6 months with or without catheter angiography in all patients. Approval for the study from the institutional ethical board was obtained, and after informed consent was obtained, repeat angiographic study was performed after 6 months in nine patients. One patient did not give consent for invasive follow-up study. Restenosis was defined by the presence of more than 50% restenosis on a follow-up angiogram. A cutting balloon 5 7 mm in diameter was passed over a inch guide wire and positioned across the stenosis. Two to three dilations with a cutting balloon at 6 8 atm for seconds were used to dilate the restenosis. The size of the cutting balloon was selected according to the diameter of the adjacent normal vessel. The cutting balloon was rotated between dilations. RESULTS Fourteen patients had severe involvement of arch arteries on aortograms. Four patients had total occlusion of all four arch vessels. These patients were sent for bypass graft surgery, as distal vessels are often patent in these patients despite proximal occlusion. Ten patients had total occlusion of three arch vessels along with a severe stenotic lesion in the only patent supraaortic vessel (ie, carotid, vertebral, or innominate). These patients were treated by stent-supported angioplasty and were prospectively followed up. Angioplasty was attempted on 12 lesions: carotid (n 8; 66.6%), vertebral (n 2; 16.6%), and brachiocephalic (n 2; 16.6%). Two patients who underwent brachiocephalic artery angioplasty also underwent carotid angioplasty (n 1) and vertebral artery dilation (n 1). Eight lesions were discrete (ie, 2 cm) and four were diffuse (ie, 2 cm). A peripheral cutting balloon was used in four cases, and balloon inflation at high pressure

3 Volume 19 Number 12 Tyagi et al 1701 ( 10 atm) was used for initial dilation in eight lesions. Patients who underwent cutting balloon angioplasty had better dilation with less dissection at lower pressure (6 8 atm) compared with balloon angioplasty. All lesions could be successfully dilated, with a variable degree of intimal tearing at the site of angioplasty. Stent implantation was performed in all patients, as the artery that had been treated with angioplasty was the sole major supply to the brain. Balloon-expandable stents were implanted in nine short-segment/ostial lesions, and self-expandable nitinol stents were implanted in three long-segment nonostial lesions. After angioplasty and stent implantation, the minimal luminal diameter increased from 0.84 mm 0.6 to 5.6 mm 0.7 (P.001), and the stenosis diameter was reduced from 81.1% 6.8% to 1.7% 3.3% (P.001; Figs 1,2). There were no procedure-related complications. No transient or permanent neurologic deficits occurred. All patients had relief of symptoms after successful angioplasty. During follow-up of 3 49 months (mean, 25 months 7), Doppler US was performed in all patients. It showed restenosis in two patients. Repeat angiographic study was performed in nine patients. One patient did not give consent for invasive follow-up study. Mild intimal hyperplasia was seen in all five patients with discrete stenosis at the time of angioplasty. Two of four patients with diffuse lesions (50%) developed restenosis. These two patients had recurrence of symptoms in the form of dizziness after 1 year. Both these patients were successfully treated with cutting balloon angioplasty. The stenosis could be effectively relieved without significant dissection with this technique. All patients continue to be free of neurologic symptoms. DISCUSSION TA is a chronic idiopathic inflammatory disease that affects large and medium-sized arteries. Clinical features reflect limb or organ ischemia resulting from stenosis or obliteration of the involved arteries. The inflammatory stage of the disease is treated with corticosteroids with the aim to arrest progression of existing lesions and prevent new lesions (11). In most Figure 1. (a) Selective angiogram of the brachiocephalic artery shows severe stenosis of the brachiocephalic artery and the origin of the right vertebral artery. The right carotid artery has long severe diffuse stenosis. Arch aortogram in this patient shows total occlusion of the left carotid and subclavian arteries. (b) Selective angiogram of brachiocephalic artery after angioplasty and stent implantation shows marked improvement in the lumen of the brachiocephalic and vertebral arteries. The patient had relief from recurrent syncope. Figure 2. (a) Selective angiogram shows severe long-segment stenosis of the right common carotid artery. The patient s vertebral, left common carotid, and left subclavian arteries were totally occluded, and the patient had recurrent syncope. (b) Angiogram shows marked improvement of the carotid artery lumen after angioplasty and selfexpandable nitinol stent placement. His symptoms were also relieved. patients, who present with chronicstage disease with severe stenosis and organ ischemia, revascularization is required (12). Lesions are often multifocal and often involve the ostium, producing irregularity, stenosis, or even total occlusion. Unlike atherosclerotic lesions, the vessels are firm, scarred, nonulcerated, and fibrotic (7). Carotid artery involvement in TA is common (6% 69%), with the left side more commonly involved than the right (13). Lupi-Herrera et al (14) showed that 6.8% of these patients developed hemiplegia and 4.5% had loss of vision at a mean follow-up of 33.2 months 37 (14). Cerebrovascular events contribute to 20% of the cases of mortality in these patients. Carotid artery bypass graft creation in this disease is associated with high rates of morbidity, mortality, cranial nerve injury, anastomotic narrowing, graft occlusion, and aneurysm formation. There may also be sudden increases in intracranial blood pressure after ca-

4 1702 Revascularization of Arch Artery for Ischemia in Takayasu Arteritis December 2008 JVIR rotid reconstruction, which may lead to cerebral hemorrhage. Severe and risky postoperative cerebral edema or ischemic reperfusion syndrome is encountered in approximately 10% of patients who are treated with surgical revascularization (12). Carotid angioplasty and stent placement are being evaluated in the treatment of carotid artery stenosis. However, most data have come from patients with atherosclerotic carotid artery involvement (15,16). The experience with carotid angioplasty in TA is limited (6 9,13,17). Our experience shows that carotid angioplasty and stent placement in patients with TA can be performed with excellent immediate and intermediateterm results. The excellent immediate results in this series could be a result of case selection with the exclusion of cases of total occlusion along with the use of cutting balloons and highpressure balloons for predilation of stenotic lesions (5). A majority of lesions dilated (75%) were short-segment lesions. The best results were obtained in short focal lesions. Long lesions tend to have incomplete dilation and are associated with a higher rate of restenosis (7). Angioplasty of a chronically occluded carotid artery is associated with a low initial success rate and high chances of restenosis. In TA, higher balloon inflation pressures are often required for dilation as a result of transmural fibrosis. In our limited experience, dilation with a cutting balloon proved to be safe and effective for initial dilation and treatment of in-stent restenosis. Balloons on the blade may help to overcome the hoop stress, leading to better dilation at lower pressures. Cerebral protection devices are not required in these nonatherosclerotic fibrotic lesions because atheroembolization is unlikely (5). Balloon-expandable stents were preferred in the present series because of their high radial strength and positioning ability, as these stenoses often involve the ostium. Self-expandable nitinol stents were preferred for long lesions away from the ostium. As the stenotic artery was the sole major supply to the brain, stents were used to enhance the safety of the procedure, which is analogous to left main coronary artery angioplasty. Balloon inflation was performed for a short duration (10 15 seconds), as longer inflation times may lead to convulsions resulting from cerebral ischemia, as other arch vessels are occluded. In aortic arteritis, stenosis of the extracranial vertebral artery is not infrequent. Kerr et al (10) reported incidences as high as 20% 40% in certain patient populations. There is a clear relationship between vertebral artery insufficiency and posterior circulation ischemia, but this is not commonly recognized because most patients can tolerate occlusion of one of the two vertebral arteries (18,19). The clinical significance of vertebral artery stenosis in aortoarteritis is underestimated in clinical practice. Surgical revascularization of an atherosclerotic vertebral artery is associated with relatively high rates of mortality (4.2%) and complications (10% 20%) such as nerve injury, as access to its origin is difficult, and pulmonary problems occur as a result of thoracotomy (20). Percutaneous transluminal angioplasty has emerged as an effective, safer therapeutic alternative to operative revascularization of proximal vertebral artery stenosis. To date there are approximately 150 reported cases of endovascular treatment with primary stent placement for extracranial vertebral artery stenosis. Most of these cases were atherosclerotic, and only few cases involved TA (8,21). These early reports have shown high technical success rates with incidences of complications (eg, minor stroke or transient ischemic attack) less than 3% and no related deaths during short-term follow-up. Angiography of the posterior and anterior circulation is required before angioplasty is performed. Special attention must also be paid to collateral supply from the contralateral vertebral artery, as well as the carotid artery, from which collateral vessels may reconstitute the distal vertebral artery in cases of vertebral occlusion or severe stenosis. The presence of bilateral disease and diffuse disease in arch vessels is associated with a higher risk of neurologic complications. Late presentation permitted palliative endovascular revascularization. Detection of cases at an earlier stage of disease ie, before total occlusion occurs could have enabled more complete revascularization by angioplasty of other stenotic arteries before the occlusive phase. The present series describes patients with TA with severe cerebrovascular symptoms in whom angioplasty and primary stent placement were performed to treat severe arch vessel stenosis. Although the present analysis is limited by its retrospective nature, small number of study subjects, and relatively short follow-up period, it shows that endoluminal stent placement of carotid and vertebral artery lesions is safe and effective and produces marked relief of symptoms and durable results, as evidenced by the low recurrence rate. It may be a suitable alternative to surgery in a select subgroup of patients, as it is associated with lower morbidity rates, good success rates, and shorter periods of hospitalization. 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