Neuroradiology (2005) 47: 282286 DOI 10.

1007/s00234-005-1343-2

INTERVENTIONAL NEURORADIOLOGY

Meritxell Garcia Alzamora Steen K. Rosahl Jens Lehmberg Joachim Klisch

Life-threatening bleeding from a vertebral artery pseudoaneurysm after anterior cervical spine approach: endovascular repair by a triple stent-in-stent method. Case report

Received: 16 August 2004 Accepted: 16 November 2004 Published online: 24 March 2005 Springer-Verlag 2005

M. Garcia Alzamora (&) J. Klisch Section of Neuroradiology, Neurocenter, University of Freiburg, Breisacher Str. 64, 79106 Freiburg, Germany E-mail: [email protected] Tel.: +49-761-2705180 S. K. Rosahl J. Lehmberg Department of Neurosurgery, Neurocenter, University of Freiburg, Breisacher Str. 64, 79106 Freiburg, Germany

Abstract The incidence of injury to the cervical vertebral artery during surgery for stenosis of the cervical neuroforamina is very low. We present a case in which bleeding during microforaminotomy at the level C6/7 occurred. The bleeding could be controlled intraoperatively. Two days later, a life-threatening cervical hematoma required urgent bedside evacuation. A false aneurysm of the left cervical vertebral artery was successfully occluded by a modied triple stent-in-stent technique, maintaining the ow in the vessel.

Keywords Cervical disk disease Anterior cervical approach Surgical injury to the cervical vertebral artery Angiography Pseudoaneurysm Vessel preservation by endovascular triple stent placement

Introduction
The anterior approach to the cervical spine is widely used in the surgical treatment of degenerative cervical spine disease. It is generally considered to be safe and eective. Due to its anatomical relationship adjacent to the neuroforamen, however, the vertebral artery is at risk during foraminotomy. The formation of a cervical vertebral artery pseudoaneurysm leading to potentially lethal situations has been reported previously [1]. Vascular complications of the anterior approach to the cervical spine also include the formation of arteriovenous stulae, late-onset hemorrhages and thrombosis with embolic incidents [2]. There are no established management strategies for suspected postoperative complications concerning the extracranial part of the vertebral artery after surgical anterior approach. Since clinical symptoms might follow vessel laceration with considerable delay, life-threatening situations due to vessel injury can occur several days up to years after surgery [3]. While diagnostic angiography can detect

the majority of these vascular complications, there has been no consensus regarding its indication and timing until now. Reported treatment strategies for vertebral artery injury include intraoperative surgical repair, vessel ligation, coil embolization, stent-assisted coil embolization and the use of stent grafts or covered stents [4]. We report on a patient in whom a pseudoaneurysm of the cervical vertebral artery following foraminotomy at the level C6/7 was treated by a modied triple stent-in-stent technique achieving aneurysm obliteration with preservation of the cervical vertebral artery.

Case report
A 50-year-old woman was admitted with a history of severe left-sided cervicobrachialgia irradiating along the C7 distribution to the second, third and fourth nger. Radiculopathy had resulted in sensory loss in the C7 dermatome. The pain was exacerbating and unresponsive

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to analgesics. Myelography and postmyelographic CT scan demonstrated compression of the left C7 nerve root both by soft tissue and spondylosis narrowing the C6/7 neuroforamen (Fig. 1a,b). In addition, anaesthetic block of the C7 nerve root resulted in immediate, albeit temporary pain relief. Via an anterior approach the intervertebral disk at the level C6/7 was removed. While the left neuroforamen was opened with a diamond drill, arterial hemorrhage occurred, that could be controlled by surgical packing with gelfoam. The intervertebral space was partially lled with articial bone cement to accomplish ventral cervical fusion. Immediate postoperative care was uneventful; the patient did not show any focal-neurological decits. She reported complete relief from her preoperative complaints.

Fig. 1 a Cervical myelogram, p.a. view revealing a C7 nerve root compression on the left side. b Post-myelo CT scan demonstrating a left foraminal stenosis at the C6/7 level. c Angiogram of the left ascending cervical artery demonstrating an active bleeding of a muscular branch on the right side after bedside evacuation of the neck hematoma. d Angiogram of the left vertebral artery, p.a. view. Laceration of the vertebral artery resulting in a false aneurysm extending medially into the intervertebral space. Note that there is no surrounding material within the former disk space, which will stabilize the vessel during stenting. To achieve stabilization of the vessel wall, the length of the stent should cover the whole cervical segment in order to minimize the risk of vessel rupture during balloon ination

Twenty-four hours later, the patient developed shortness of breath and asphyxia requiring immediate bedside evacuation of a hematoma of the soft tissue of the neck followed by emergency tracheostomy and intubation. After stabilization of the patients vital parameters, angiography was performed. It revealed active bleeding from a muscular branch on the right side which probably had been aected during bedside evacuation of the hematoma. The bleeding was stopped by transarterial embolization with liquid embolic (Fig. 1c). While injecting the left vertebral artery, a false aneurysm of the vessel at the operated level C6/7 was found, extending medially into the intervertebral space (Fig. 1d). All attempts to obtain direct access through the small laceration of the vessel by the use of dierent microguidewires and microcatheters failed. After measuring the vessel diameter, a 25-mm long balloonexpandable stent of 3.5 mm diameter (AVE Cerebrence over-the-wire; Medtronic Inc.) was positioned using 6 atm ination pressure to achieve stabilization within the vessel from the superior end plate of C6 to the inferior end plate of C7 (Fig. 2a, b). A second stent of same diameter with a length of 9 mm (AVE Cerebrence over-the-wire; Medtronic Inc.) was deployed within the rst stent exactly over the laceration of the vertebral artery. Over a period of 20 min, control series showed reduced but persistent lling of the aneurysm (Fig. 2c, d). Therefore, it was decided to introduce a third uncovered stent of 7 mm in length and 3.5 mm in

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Fig. 2 a Subtracted and b unsubtracted angiogram of the left vertebral artery after the rst stent was deployed (3.5 mm diameter, 25 mm length, 6 atm, AVE Cerebrence over-the-wire; Medtronic Inc.). c Subtracted and d unsubtracted angiogram of the left vertebral artery after deployment of the second stent (3.5 mm diameter, 9 mm length, 6 atm, AVE Cerebrence over-the-wire; Medtronic Inc.). Note that the second stent is placed within the center of the rst one, yet still there was persistent lling of the aneurysm. e Subtracted and f unsubtracted angiogram of the left vertebral artery after deployment of the third stent (3.5 mm diameter, 7 mm length, 7 atm, AVE Cerebrence over-the-wire; Medtronic, Inc.). Complete occlusion of the false aneurysm. Note that the third stent is exactly located within the second one covering the laceration of the vessel wall (see also g). g CT-scan C6/7 (bonewindow) after endovascular repair of the left vertebral artery demonstrating the position of the triple stent segment close to the opened neuroforamen. Note that contrast lling of the false aneurysm persisted within the intervertebral disc space after closure of the vertebral artery laceration

After closure of the aneurysm, anticoagulation was started using heparin up to 1.5 times the normal activated PTT dose. Aspirin medication was started 12 h after intervention.

Discussion
Vascular complications during anterior approaches to the cervical spine are rare ($0.3%), but can potentially lead to serious morbidity and mortality [1]. Complications include arteriovenous stulae, late-onset bleeding, and pseudoaneurysms. There are only a few reports about vascular complications in anterior cervical spine surgery [2]. Due to their high rate of rupture and rebleeding in the unsecured state, prompt recognition and treatment of pseudoaneurysms is of utmost importance to avoid life-threatening situations. As vessel injury may be clinically obscured, observation of the patient may not suce in this situation. Bleeding may occur at variable intervals from the time of injury, rendering prompt diagnosis of false vertebral artery aneurysm clinically challenging. Urgent evaluation of the vertebral vessel status is indicated when severe arterial bleeding is encountered during surgery [2]. The question arises whether angiography should be performed in cases of minor arterial hemorrhage which was easily controlled intraoperatively. On the other hand, a normal angiography after surgery complicated by vertebral vessel injury, does not rule out later formation of a false aneurysm, since rebleeding days to years after surgery has been described [3]. The role and signicance of

diameter (AVE Cerebrence over-the-wire; Medtronic Inc.) within the previous two stents (Fig. 2e, f). Similar to the rst stent, the second and third stents were applied using 6 atm ination pressure. After exact placement, the balloon was inated up to 7 atm pressure to obtain optimal compression and narrowing of the stents facilitating obliteration of the vessel laceration. After the third stent was in place, the aneurysm was completely excluded with preservation of orthograde perfusion of the vertebral artery. Thirty minutes as well as 24 h later, control series excluded a residual aneurysm and demonstrated an intact and patent cervical vertebral artery. CT scan showed the exact localization of the triple stent at the level of C6/7 (Fig. 2g).

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repeated angiography has not yet been assessed. It is suggested that in cases suggestive of vessel laceration, patients should be followed up by MR imaging to evaluate the vessel situation and to exclude a growing false aneurysm. This situation is similar to transphenoidal surgery, if iatrogenic injury to the carotid artery has occurred. Postoperative control angiography in this setting has been strongly recommended and has become a standard in many centers [5]. If intraoperative profuse bleeding occurs, immediate intra-operative control of the hemorrhage is urgent and is generally obtained by means of compressive packing. With respect to the present case, we think that postoperative angiography should be indicated generously in order to avoid life threatening bleeding. For the repair of vertebral artery lesions, the following procedures have been proposed: intraoperative surgical repair [6], postoperative surgical treatment including ligation, vessel occlusion with or without vascular reconstruction, trapping, bypass procedures, clipping or the use of stent grafts [4]. These procedures are technically demanding, invasive and have a high morbidity rate. In addition, dierent endovascular treatment options like endovascular occlusion [7], stent supported coil embolization [8], stent assisted angioplasty, the application of balloons or coils have been described [79]. Most of these procedures result in occlusion of the vessel and the outcome is dependent on adequate perfusion of the contralateral vertebral artery. Coil embolization, the triple uncovered stent-in-stent technique described above, covered stents or stent grafts may achieve immediate aneurysm occlusion with preservation of the vessel [4]. Using the stent method, both covered [4, 8] and uncovered stents may be employed. So far, there have been no long-term results in favor of one method or the other. Nevertheless, covered stents are more dicult to navigate and less available than uncovered stents. In addition, poor short term patency rates and insucient promotion of aneurysm thrombosis formation of covered stents have been described. In our case, the contralateral vertebral artery was hypoplastic, hence vessel repair with patency of the artery was desirable. Coiling was not successful because of the very small laceration of the vessel wall which was not eligible for microcatheter placement. Multiple stents have been applied to selectively eliminate the aneurysm while preserving the patency of the vertebral artery. The use of multiple stents minimizes porosity and may provide a matrix for endothelial growth causing local cell proliferation and migration [10, 11]. This regional remodeling can promote selective thrombosis of the aneurysm. A similar stent-in-stent technique is known from the cardiological literature [12] and has also been described to have succeeded in the treatment of intracranial aneurysms [10, 11] as well as in vessel dissections and stulae. In the literature, very few cases have been reported in which

pseudoaneurysms have been treated by selective stent reconstruction [2]. A triple stent-in-stent technique has never been reported for iatrogenic aneurysms of the extracranial vertebral artery. There is no doubt that this procedure carries a high risk of morbidity and mortality. Exact measurement of the diameter of the vessel is required, assuming that oversizing is not well tolerated. All three stents in the present case were deployed by the use of 6 atm ination pressure to reach 3.5 mm diameter exactly. At the end of the procedure, the triple stent package was expanded with 7 atm to compress the gaps between the stents and to maintain full patency of the vertebral artery. The absence of a trust bearing within the operated segment C6/7 did not assure stability of a single stent of 7 mm or 9 mm in length. In combination with a balloon pressure of 6 atm these factors are prone to induce vessel wall instability which in turn can lead to its rupture. The rst stent should probably be oversized in length to ensure sucient stability within the injured vessel. The risk of vessel rupture during this endovascular procedure has to be estimated to be higher as compared to stent assisted therapy of dissected aneurysms [8, 9] or acutely ruptured wide-neck aneurysms [10]. The orientation of the stent interstices cannot be adjusted because it is impossible to rotate the stent during placement and the interstices are not visible prior to and during stent release. This may have been the reason for persistent lling of the aneurysm after deployment of the second stent in our case. As stated by Doerer et al. [10], usually this is not a problem in smaller aneurysms. In contrast to small wide necked aneurysms which can be treated by the double-stent method [10, 11], persistent lling of the pseudoaneurysm in our case could not be accepted, since the risk of rebleeding was estimated as too high. Therefore, stenting was continued until no lling of the aneurysm was observed. The risk of acute stent thrombosis is high in this situation, and we assume that strong anticoagulation is not applicable. Retrospectively, anti-platelet medication (with aspirin and plavix) should probably be started immediately after apparently successful reconstruction of the vessel wall. Early and careful follow-up angiography studies are warranted to identify possible changes in pseudoaneurysm lling and to add further endovascular therapy if, for example, intimal hyperplasia should lead to luminal stenosis. On the other hand, there was no increased incidence of intimal hyperplasia in patients treated with the double stent method as compared to single stent methods. However, stents as foreign bodies promote vessel inammation. Long-term eects of uncovered stenting still have to be evaluated. Myointimal hyperplasia and future stent stenosis remain a concern for possible future hemodynamic complications, although the risk of signicant stenosis in vessels with larger diameters like the vertebral artery is much lower than in

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smaller ones [11]. The risk of perforation and ischemia is not yet suciently dened. Despite these diculties, the use of stents in vessel repair has major advantages. The technique is fast, which is useful in urgent situations as described in our case. Stenting can be combined with coiling [8, 9]. Even if stent placement fails, coil occlusion of the parent vessel can still be considered. In conclusion, we can say that there is a risk, albeit small, of formation of iatrogenic aneurysms after

microforaminotomy. Life-threatening situations with potentially devastating complications should be expeditiously recognized and managed. We have demonstrated that an endovascular multiple stent-in-stent reconstruction for a postoperative false aneurysm is a useful technical adjunct in interventional treatment strategies. It has the potential to assure vertebral artery patency. We propose the use of uncovered stents in this modied technique due to their applicability and ready availability in an emergency situation when vessel patency is warranted.

References
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