CASE REPORT

Reocclusion and stroke due to immediate plaque protrusionfollowing endovascular treatment of carotid artery successfullytreated with intra-arterial urokinase and stent in stent in a patientwith Takayasu arteritis with severe disease of all arch vessels

B. C. Srinivas • Soumya Patra • Babu Reddy •

C. M. Nagesh • C. N. Manjunath

Received: 9 January 2013 / Accepted: 27 March 2013

� Japanese Association of Cardiovascular Intervention and Therapeutics 2013

Abstract Endovascular treatment is becoming a safe and

efficacious modality in the management of carotid artery

stenosis in Takayasu arteritis (TA). A 24-year-old woman

of TA presented with blurred vision, recurrent syncope and

upper limb claudication. Angiography revealed right CCA

95 %, left CCA 90 % stenosis and occlusion of other arch

vessels. She underwent right CCA angioplasty and stent-

ing. She developed left-sided hemi paresis. Check angio-

gram revealed plaque protrusion in the proximal part of the

stented segment. Intra-arterial urokinase bolus was given

and overlapping stenting done. Re-occlusion by plaque

protrusion immediately after stenting like in our case is not

reported.

Keywords Carotid artery � Endovascular treatment �Intra-arterial urokinase � Plaque protrusion � Takayasu

arteritis

Introduction

Takayasu arteritis (TA) is a chronic inflammatory disease

affecting the aorta and its major branches. The most fre-

quently involved vessels are subclavian arteries, carotid

arteries, descending thoracic or abdominal aorta, and renal

arteries [1]. TA primarily affects young women of child-

bearing age and usually presents with absent radial pulse

and signs of cerebral ischemia [1]. Carotid angioplasty and

stenting are increasingly being used as safe and efficacious

modality of treatment in the management of carotid artery

stenosis [2]. Restenosis rates of percutaneously treated

Takayasu lesions have been higher than those with non-

inflammatory lesions. But so far medline search does not

reveal any case before where patient had post-procedure

stroke due to immediate occlusion of stent by plaque

protrusion.

Case report

A 24-year-old woman presented with progressive blurring

of vision, recurrent episodes of syncope and bilateral upper

limb claudication for last 1 year. There was no significant

family history. On general examination bilateral upper

limb pulse was not palpable. But, the lower limb pulses

were palpable and blood pressure (BP) recorded at lower

limb showed 160/85. On cardiovascular system examina-

tion, there was loud first heart sound and bilateral carotid

artery bruit. Neurological and other system examination

did not reveal any significant abnormality. There were

ophthalmologic features of hypertensive retinopathy. As

she was a young female and had pulselessness in bilateral

upper limb with hypertension and carotid artery bruit, TA

was diagnosed clinically in our case. Blood investigations

showed presence of anemia (hemoglobin, 10.2 gm/dl),

leucocytosis (12500/mm3) and high erythrocyte sedimen-

tation rate (ESR, 56 mm). Renal and liver function tests

showed normal results. She was on regular treatment with

oral methotrexate because she had history of steroid tox-

icity at the beginning when it was started after diagnosis of

Electronic supplementary material The online version of thisarticle (doi:10.1007/s12928-013-0176-x) contains supplementarymaterial, which is available to authorized users.

B. C. Srinivas � S. Patra (&) � B. Reddy �C. M. Nagesh � C. N. Manjunath

Department of Cardiology, Sri Jayadeva Institute

of Cardiovascular Sciences and Research,

Bangalore 560069, Karnataka, India

e-mail: dr_soumyapatra@rediffmail.com

123

Cardiovasc Interv and Ther

DOI 10.1007/s12928-013-0176-x

TA. CT aortogram and arch vessel were done which

showed significant stenosis of B/L common carotid artery

(CCA) and total occlusion of B/L subclavian artery (SCA),

but there was no evidence of high plaque burden seen.

Angiography revealed the same with right CCA 99 %, left

CCA 90 % stenosis and occlusion of other arch vessels

(Fig. 1). As our patient had progressive loss of vision, plan

was to go ahead with endovascular treatment of CCA and

as right CCA had more stenosis than left CCA, angioplasty

and stenting of right CCA were planned at first sitting. She

was pre-medicated with dual antiplatelet therapy (DAPT)

and loading dose of aspirin and clopidogrel was given on

the day before the procedure and also on the day of the

procedure. During the procedure, unfractionated heparin

(UFH) bolus (5000 units) was given at the beginning. She

was stented successfully and uneventfully by using long

7 9 120 mm smart precise cordis stent (Fig. 2). Distal

embolic protection device was not used in our case because

TA is not a disease with high plaque burden like that in

atheromatous carotid artery stenosis and she could not

afford that also. Immediately after the procedure activated

clotting time (ACT) was 290 s. During observation in post-

catheterisation word, she developed left-sided hemiparesis

within 45 min of the procedure. She was brought back to

catheterisation laboratory and check angiogram taken

which showed occlusion of the stent proximally by plaque

protrusion (Fig. 3). Cerebral angiogram of brain (Fig. 4)

was done immediately which showed embolic occlusion of

inferior division of right middle cerebral artery (MCA). For

the reason of acute stroke along with plaque protrusion

which was thought to be the reason for stroke in our

patient, she was managed immediately by intra-arterially

administered bolus of 2 lacks unit urokinase. Post-proce-

dure she had residual weakness and CT brain also showed

large infarct in the right MCA territory. She was re-stented

by a same overlapping stent (Fig. 5) as there was no

available close-cell stent in our catheterization laboratory.

But later she improved neurologically and was followed up

over the last 6 months. Now, she has good vision, no

syncope and only minimal weakness of left side of the

body (power-4?/5). She is also treated with weekly dose of

Fig. 1 Angiography revealed 99 % stenosis of right CCA and total

occlusion of right SCA

Fig. 2 Stented successfully by using long 7 9 120 mm smart precise

cordis stent

Fig. 3 Check angiogram showed occlusion of the stent proximally by

plaque protrusion

B. C. Srinivas et al.

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oral methotrexate, folinic acid and DAPT with aspirin

(150 mg/day) and clopidogrel (75 mg/day).

Discussion

In our case, the patient presented mainly with neurological

symptoms like blurring of vision and recurrent syncopal

attack. The neurological manifestations of TA result pri-

marily from decreased blood flow caused by steno-occlu-

sive lesions in the arch and cervical arteries to the brain

and/or shifting of the blood flow (steal) [3]. Neurological

manifestations are most often associated with obstructive

lesions of multiple arch and cervical arteries, occurring

during the latest stages of disease progression which have

been reported in 57 to 80 % of patients, may include

headache, dizziness, visual disturbance or loss, stroke, and

transient ischemic attack (TIA) [3]. Management strategies

for TA include medical therapy with steroids or immuno-

suppressive agents and revascularization procedures. In

conjunction with medical treatment, PTA and/or stenting of

supra-aortic arteries in TA is feasible and durable, and

provides good symptomatic relief in patients with multiple

stenoocclusive lesions of supra-aortic arteries [4]. Stent-

supported angioplasty of a sole supraaortic artery in TA is

safe and effective and provides good symptomatic relief in

patients with multiple steno-occlusive lesions of arch

arteries like in our case [2]. Although surgical treatment

has been used to bypass the stenosed segment, the diffuse,

proximal, and multifocal involvement of the arch vessels

may make surgical revascularization difficult [3]. Endo-

vascular treatment has emerged as the initial mode of

treatment for stenosis of the aorta, renal, and subclavian

arteries caused by TA. Less is known, however, about the

role of endovascular treatment for lesions in the carotid,

brachiocephalic, and vertebral arteries [5]. Progressive loss

of vision was the reason behind to stent right CCA at the

first step in our case. Carotid artery stenting (CAS) place-

ment was shown to be a feasible option for treating long

segment stenosis of carotid arteries in patients with TA

with encouraging results [6] and in our case, we have used

long 120 mm stent. There have been some reports of re-

occlusion due to plaque protrusion after CAS for athero-

sclerotic stenosis but this is rarely seen in TA as in TA

carotid artery stenosis is mainly due to fibrotic occlusion of

vascular lumen and even though the ACT was in thera-

peutic range [6]. Restenosis rates of percutaneously treated

Takayasu lesions are high and especially diffuse lesions of

arch vessels are associated with a higher rate of restenosis

than focal lesions [7]. Though, CT angiogram didn’t reveal

any features of plaque burden, but MRI or intravascular

ultrasound (IVS) are considered as better mode of inves-

tigations for this purpose.

As our patient had acute stroke with occlusion of MCA

territory, we treated this case with intra-arterial urokinase.

At that moment, she was very sick so we did not take much

time for restenting after intra-arterial injection. Our patient

improved dramatically on the very next day which indi-

rectly suggests that our treatment with lytic therapy was

effective. It might be better if we could demonstrate one

cerebral angiogram after lysis showing effective lysis but at

that situation our primary aim was to address the plaque

and clot burden within the carotid artery which was

threatening to be embolized again by plastering them

against the vessel wall by another stent as soon as possible

and we were very much successful.

Fig. 4 Cerebral angiogram (lateral view) showed embolic occlusion

of inferior division of right MCA (black arrow)

Fig. 5 Re-stented by an overlapping stent

Stroke and reocclusion following CAS in TA

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Because ESR at the time of admission may affect the

results of endovascular treatment, it is important to strictly

control active disease by administering immunosuppressive

agents before and after endovascular treatment [8]. Though

we had treated our case with oral methotrexate, admission

ESR was high and it might be the reason for immediate

plaque protrusion in our case as it is mandatory to control

inflammation before performing any intervention in TA.

We should keep in mind that endovascular stenting for TA

does not always keep the patency of the affected vessels,

and severity of the stenosis and/or uncontrollable systemic

inflammation could be a risk factor for restenosis. There-

fore, careful follow-up under strict control of inflammation

is mandatory. In a case of immediate stent closure by

plaque protrusion, closed-cell design stent seems to be

preferable for stenting as what we should do in our case.

But, in our catheterization laboratory, there was no avail-

ability of closed-cell design stent. Novel approaches are

being sought to treat restenosis in these patients, such as

the use of stent grafts [9]. Though reopening a chronically

occluded carotid artery also risks an embolic shower to the

brain, immediate post-procedural stroke due to occlusion of

stent by plaque protrusion is unknown. Using multiple sites

of arterial access, distal embolic protection devices can be

deployed in both the vertebral and internal carotid arteries

to reduce the risk of stroke [10] what we did not use in our

case.

Conclusion

Endovascular treatment with stenting with a long stent is

feasible in maintaining patency of carotid artery in TA.

Before percutaneous intervention, disease activity should

be controlled. Distal embolic protection should be used if

feasible in these cases. We should observe carefully during

post-procedural period for the development of complica-

tion (stroke, etc.) like in our case and if it is so, then it can

be managed successfully by administering immediate intra-

arterial fibrinolytic therapy followed by an overlapping

stenting of carotid artery and especially by a stent which

has closed-cell design.

Conflict of interest None.

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