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Clinical case

Vertebral artery (V2) pseudo-aneurysm after surgery for cervical schwannoma. How to prevent it and a review of the literature Pseudo-anévrisme de l’artère vertébrale (segment V2) au décours d’une chirurgie de schwannome cervical. Comment prévenir cette complication, revue de la littérature V. Jecko a,∗ , M. Rué a , V. Castetbon b , J. Berge c , J.-R. Vignes a a b c

Neurosurgery A Unit, University of Bordeaux-Segalen, Pellegrin University Hospital, Bordeaux, France ENT Unit, University of Bordeaux-Segalen, Pellegrin University Hospital, Bordeaux, France Neuroradiology Unit, University of Bordeaux-Segalen, Pellegrin University Hospital, Bordeaux, France

a r t i c l e

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Article history: Received 7 April 2014 Received in revised form 22 July 2014 Accepted 28 August 2014 Available online xxx Keywords: Vertebral artery Anterior approach Pseudo-aneurysm Schwannoma Endovascular treatment Complication Prevention Surgical planning Cervical spine

a b s t r a c t Study design. – Case report and review of the literature. Objective. – To prevent and manage a suspected iatrogenic vertebral artery injury during a cervical spine anterior approach. Summary of background data. – The anterior spine approach is a common surgery with few complications. One of the rare but significant risks is vertebral artery injury. Consequences of vertebral artery injuries are often delayed. Therefore, it is essential to prevent this complication and to know how when exploring after a suspected vertebral artery injury. Methods. – Report of a case and review of the literature. A 61-year-old woman presented with a cervical schwannoma involving the C5–C6 foramen. She had undergone surgery 22 years before by the posterior approach. We performed an anterior cervical approach. After 12 days, a vertebral artery pseudo-aneurysm occurred. Our review of the literature is focalized on vertebral artery injuries during cervical surgery by the anterior approach. Results. – The patient was treated by coil embolization with a good outcome. To our knowledge, only 6 cases of vertebral artery pseudo-aneurysm after surgery have been reported in the literature. Conclusion. – According to the literature, vertebral artery pseudo-aneurysms resulting in anterior cervical approach are rare but their consequences could be severe. Prevention begins by detailed surgical planning. Peroperative imaging is helpful. Any suspected vertebral artery injury should postpone a contralateral approach before angiographic imaging. © 2014 Elsevier Masson SAS. All rights reserved.

r é s u m é Mots clés : Artère vertébrale Abord cervical antérieur Pseudo-anévrisme Schwannome Traitement endovasculaire Complication Prévention Planning chirurgical Rachis cervical

Type d’étude. – Étude de cas et revue de la littérature. Objectif. – Prévenir et prendre en charge les traumatismes supposés de l’artère vertébrale survenant au décours des abords du rachis cervical par voie antérieure. Données existantes. – Les abords antérieurs du rachis cervical sont connus pour leur faible taux de complications. Parmi elles, les traumatismes de l’artère vertébrale sont parmi les plus rares mais peuvent avoir de lourdes conséquences, le plus souvent survenant secondairement. Il est donc essentiel de prévenir ce type de complication et de savoir comment compléter le bilan en cas de suspicion de traumatisme de l’artère vertébrale. Méthodes. – Nous rapportons le cas d’une patiente de 61 ans, présentant un neurinome foraminal C5–C6 droit. Elle avait déjà subi une chirurgie, 22 ans auparavant, par abord cervical postérieur. Un abord

∗ Corresponding author. Service de neurochirurgie A, CHU de Bordeaux, place Amélie-Raba-Léon, 33076 Bordeaux cedex, France. E-mail address: [email protected] (V. Jecko). http://dx.doi.org/10.1016/j.neuchi.2014.08.002 0028-3770/© 2014 Elsevier Masson SAS. All rights reserved.

Please cite this article in press as: Jecko V, et al. Vertebral artery (V2) pseudo-aneurysm after surgery for cervical schwannoma. How to prevent it and a review of the literature. Neurochirurgie (2015), http://dx.doi.org/10.1016/j.neuchi.2014.08.002

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antérieur a été réalisé. Douze jours après la chirurgie, la patiente a présenté une rupture d’un anévrisme de l’artère vertébrale droite. Nous avons confronté nos résultats à ceux de la littérature rapportant des traumatismes de l’artère vertébrale au cours des abords cervicaux antérieurs. Résultats. – La patiente a été traitée par embolisation de l’artère vertébrale droite ; les suites ont été simples. Seulement 6 cas d’anévrismes de l’artère vertébrale sont rapportés dans la littérature chez des patients ayant subi des abords antérieurs du rachis cervical. Conclusion. – Les anévrismes traumatiques de l’artère vertébrale survenant au décours de la chirurgie du rachis cervical par voie antérieure sont très rares, mais leurs conséquences peuvent être dramatiques. La prévention passe par une planification chirurgicale soigneuse étudiant le réseau vasculaire et ses suppléances. L’imagerie peropératoire pourrait être utile ; cependant, toute suspicion de traumatisme d’une artère vertébrale doit faire différer un abord controlatéral. © 2014 Elsevier Masson SAS. Tous droits réservés.

1. Introduction Vertebral artery injury during cervical spine surgery is a relatively uncommon complication. This risk is well known during C2–C1 arthrodesis, where there is up to 8.2% of lesions according to several reported series [1–6] and 0.5% during anterior cervical decompression [1,3,7,8]. Iatrogenic aneurysm associated with cervical surgery is an exceptional adverse event. We report the case of a vertebral pseudo-aneurysm, occurring a few days after the resection of a cervical schwannoma through a cervical anterolateral approach. 2. Case report A 61-year-old woman presented with a right C6 cervico-brachial neuralgia, which had developed over a period of 15 days. She had a right C6 radicular weakness for three days. Clinical examination showed right biceps reflex abolition. Cervical MRI scan performed in emergency showed an expansive process inside and outside the right C5–C6 foramen following C6 root (Fig. 1). The patient had previously undergone surgery for a C5–C6 schwannoma 22 years earlier via a posterior surgical approach.

A right anterolateral cervical spine approach was performed. The right vertebral artery was exposed laterally in its V2 portion by drilling in order to open the foramina; minor bleeding was observed, only requiring a small packing of SurgicelTM (oxidized regenerated cellulose). Debulking of the schwannoma was achieved with an ultrasonic surgical aspirator. At the end of the procedure, we performed an anterior C5–C6 fusion using screws and plates. After examination under an operating microscope, we did not observe any vertebral artery trauma before closure. Pathological examination confirmed a schwannoma (WHO grade I). The immediate postoperative course was favorable with significant regression of radicular pain and early motor recovery. The patient was discharged home five days later. Twelve days after surgery, as a result of a sudden neck movement, the patient reported feeling cervical pain. She described the appearance of a right cervical tumefaction, quickly complicated by dyspnea requiring emergency hospitalization. The patient was in hemorrhagic shock upon arrival, she was intubated and sedated. The CT scan showed right cervical and right para-pharyngeal hematoma associated with a hemomediastinum caused by rupture of a right vertebral artery pseudo-aneurysm in its V2 section (Figs. 2 and 3). The patient immediately underwent arteriography to assess the cervical spine vascularization and to visualize the dominant vertebral artery. During the same procedure, endovascular occlusion of the right vertebral artery was performed (Figs. 4 and 5). After the procedure, the patient had an ischemic stroke in the area of the right posterior inferior cerebellar artery. One year later, the patient recovered totally and she did not complain of any residual pain. 3. Discussion 3.1. Anatomical considerations

Fig. 1. MRI, axial, scalloping of the vertebral body (large black arrow) by the C6 schwannoma (small black arrow), vertebral arteries (white arrows) showing discrepant diameters. The patient underwent a posterior approach 22 years before. IRM pré-opératoire, séquence pondérée T1, coupe axiale. Scalloping du corps vertébral (grosse flèche noire) par un neurinome de la racine C6 droite (petite flèche noire). Flèches blanches montrant une différence de diamètre entre les deux artères vertébrales. La patiente avait eu une chirurgie par abord postérieur il y a 22 ans.

The vertebral artery is divided into four sections: V1, formed by the movable portion of the vertebral artery, from the subclavian artery to the transverse foramen of C6; V2, the transverse process segment fixed from C6 to C2; V3, from C2 to the penetration of the dura at the foramen magnum; and V4, the intradural segment, from the foramen magnum to the junction with the contralateral vertebral artery [9]. Concerning the V2 segment, the vertebral artery runs into the canal formed by the transverse foramen, the intervertebral muscles, the anterior scalene muscles, and the long neck muscles. When passing through this canal, the vertebral artery is surrounded by a rich venous plexus and the transverse foramen of C6 is generally oriented posteriorly and medially [3,6,10]. Inside the intertransverse canal, the vertebral artery rises closer to the lateral border of the vertebral body compared to the medial border of the anterior tubercle of the transverse process [11]. One of the main features of the cervical anterior approach is to identify the

Please cite this article in press as: Jecko V, et al. Vertebral artery (V2) pseudo-aneurysm after surgery for cervical schwannoma. How to prevent it and a review of the literature. Neurochirurgie (2015), http://dx.doi.org/10.1016/j.neuchi.2014.08.002

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Fig. 2. Angiographic CT scan; anterior view. Vertebral pseudo-aneurysm (small arrow) and anterior arthrodesis plate (large arrow). Angio-scanner cervical, reconstruction volumétrique, vue antérieure. Pseudo-anévrisme de l’artère vertébrale gauche (petite flèche) et arthrodèse antérieure par plaque vissée C5–C6 (grosse flèche).

center line [12]. That is why the longus colli muscles are important landmarks. We recommend marking the middle of the intermuscular space before starting the dissection according to Peng et al. [3]. This mark will be the reference to avoid wandering sideways. The

Fig. 3. Angiographic CT scan; postero-lateral view (right side), 12 days after surgery. Enlargement of the intervertebral foramen. Right vertebral artery pseudo-aneurysm (small arrow). Angio-scanner cervical, reconstruction volumétrique, vue postéro-latérale droite, réalisé 12 jours après la chirurgie. Élargissement du foramen intervertébral. Pseudo-anévrisme de l’artère vertébrale droite (petite flèche).

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Fig. 4. Roadmapping during right vertebral artery embolization, right vertebral pseudo-aneurysm (big arrow), microcatheter in the right vertebral artery (small arrow). Images de roadmapping au cours de l’embolisation du pseudo-anévrisme de l’artère vertébrale droite (grosse flèche) ; microcathéter situé en amont du sac anévrismal (petite flèche).

vertebral artery travels about 15 mm from the midline at C3–C4 level and 17 mm at C5–C6 [12]. The two vertebral arteries converge in their upper segments, which means it must be approached cautiously when close to the upper cervical spine [13]. When

Fig. 5. Right carotid artery angiography, final finding after right vertebral artery coiling, common carotid artery (small arrow), coils in the right vertebral artery (big arrow). Artériographie de l’artère carotide commune droite (petite flèche), contrôle final après sacrifice de l’artère vertébrale droite à l’aide de coils (grosse flèche).

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performing a vertebrectomy, most authors recommend not exceeding the edge of the vertebral body to preserve the foramen of the vertebral artery. 3.2. Anatomical variations However, nearly 20% of the population have a malposition of the vertebral artery [3], especially the V1 and V2 segments. V1 is tortuous in 22 to 47% of cases, most often with a medial loop, according to the study of Poonam et al. in 2011 [14]. Loops in the V2 segment of the vertebral artery in the transverse canal are less frequently described, which can lead to erosion of the vertebral body [15]. In 2006, Bruneau et al. analyzed the V2 segment by MRI or CT angiography. In 250 patients, they analyzed 500 vertebral arteries; they found 7% entering the transverse foramen at C6 level, with an asymmetry in 12.4% of cases [16]. These data are comparable with a radiological study finding 5.1% of vertebral arteries penetrating to another level than the C6 foramen [17]. These abnormalities of transverse canal are associated in rare cases (5.6%) with a vascular loop at the medial part of the longus colli muscle [17]. Furthermore, the size of the vertebral arteries is usually asymmetric in the same patient, with a large variation in size and position within the transverse foramen, the artery occupying 8 to 85% of the internal diameter of the foramen [18]. Our patient did not present any anatomical variation based on the usual configuration described above. 3.3. Literature data Iatrogenic injuries of the vertebral artery in the V2 segment are rare during cervical spine surgery via an anterior approach [1,3,7,8,19], only 0.5% in Tang and Rao’s series [20]. These injuries occur mainly in a corporectomy or foraminal hernia. Different types of injuries may occur: arterial wound, arteriovenous fistula, complete or incomplete thrombosis of the vertebral artery resulting in cerebral embolism, or pseudo-aneurysm of the vertebral artery. Pseudo-aneurysm usually develops after an arterial wound for which the bleeding is controlled by packing and local hemostatic agents during surgery. It can be an unnoticed trauma of the arterial wall during the operating procedure as in our case report. Cases of pseudo-aneurysm due to cervical spine surgery via an anterior approach are exceptional with only 6 cases reported in the literature [1,8,21,22]. Accurately counting these cases is difficult since the largest series do not identify the type of lesion, but focus primarily on the mechanism of its origin. Moreover, those complications appear only secondarily, therefore they are probably underestimated. Before the rupture, the diagnosis of vertebral pseudo-aneurysm can be made on the occurrence of isolated cranial nerve palsy by direct compression, on the appearance of a cervical mass, or on the onset of severe tinnitus [23]. The consequences of any type of vertebral artery injuries are highly variable and depend in particular on the side of the stroke [1,7,22]: the left vertebral artery is most often dominant [24]. Fortunately, in our case, the trauma concerned the right vertebral artery. This artery was found to be diminutive and non-dominant. Morbidity and mortality studies vary from 0% [20] to 33% [7], however, the consequences could be dramatic: hemiplegia, posterior fossa stroke, Wallenberg’s syndrome, and cranial nerve palsies. 3.4. Preventive measures Most authors recommend a careful analysis of preoperative radiological anatomy to highlight the path of the V2 segment with CT or MR angiography [10,25]. Both radiological assessments are relevant to identify the transverse process foramen, the presence of a median vascular loop at the V1 segment, the size and position

of the artery in the transverse process foramen, and the presence of bone erosion of the vertebral body due to a vascular loop in the transverse segment [16,17]. Any surgical difficulty may be anticipated. In our case, there was no variation in the path of the vertebral artery; however, the right C6 schwannoma modified the anatomy, increasing the inter-lamina space and enlarging the foramen. Moreover, our patient had never undergone surgery; this fact could have deeply modified the usual anatomy. In cases of a high risk of arterial injury, some authors advocate analyzing the circle of Willis and comparing the flow of the two vertebral arteries before surgery to identify the dominant one [7]. During surgery, anatomical landmarks such as the midline materialized by the middle part of the longus colli muscle, the lateral edge of the vertebral body, or the uncus are major markers [10–12,19]. Also, new techniques of computer-assisted surgery may be useful while drilling close to the vertebral foramen [3,26]. Some authors described this technique in cervical foraminal hernia surgery [27]. If there is any suspicion of wound or injury of the vertebral artery, a contralateral approach must be avoided due to the risk of further injury and fatal brainstem ischemia [2]. Indocyanine green video angiography could be helpful to analyze the vessels, moreover a pseudo-aneurysm only occurs over time. In any event, a simple examination under an operating microscope is insufficient. These patients must benefit an early angiographic exploration due to the delayed risk of pseudo-aneurysm rupture as illustrated in our case [1,22,28,29]. During the surgery, management of vertebral artery injury consists of hemostatic packing. This is sufficient in most cases. Ligation of the vertebral artery is an option but must only be used if bleeding is out of control because of the risk of cerebellar or brainstem infarction [7]. 4. Conclusion Iatrogenic lesions of the vertebral artery are a rare complication occurring due to failure of surgery via an anterior cervical spine approach. Occurrence of pseudo-aneurysms is very rare. However, it could be associated with major delayed complications causing morbidity and mortality. The best prevention is carefully planning the surgery using bone and vascular imaging. If a traumatic injury of the vertebral artery is suspected, an angiogram should be performed prior to discharge home and treatment must not be delayed. Disclosure of interest The authors declare that they have no conflicts of interest concerning this article. Acknowledgements The authors thank Professor Bernard George, Department of Neurosurgery, University Hospital of Lariboisière, France, for his helpful comments. References [1] Neo M, Fujibayashi S, Miyata M, Takemoto M, Nakamura T. Vertebral artery injury during cervical spine surgery. Spine 2008;33(7):779–85. [2] Wright NM, Lauryssen C, American Association of Neurological Surgeons/Congress of Neurological Surgeons. Vertebral artery injury in C1–2 transarticular screw fixation: results of a survey of the AANS/CNS section on disorders of the spine and peripheral nerves. J Neurosurg 1998;88(4):634–40. [3] Peng CW, Chou BT, Bendo JA, Spivak JM. Vertebral artery injury in cervical spine surgery: anatomical considerations, management, and preventive measures. Spine J 2009;9(1):70–6. [4] Xu H, Chi YL, Wang XY, Dou HC, Wang S, Huang YX, et al. Comparison of the anatomic risk for vertebral artery injury associated with percutaneous

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Vertebral artery (V2) pseudo-aneurysm after surgery for cervical schwannoma. How to prevent it and a review of the literature.

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