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Treatment of traumatic vertebral arteriovenous fistula by balloon occlusion and compressive manoeuvres
Eur ] VascSurg 2, 133-139 (1988)
CASE REPORT T r e a t m e n t of T r a u m a t i c Vertebral Arteriovenous Fistula by Balloon Occlusion and Compressive M a n o e u v r e s Michael Nelson*, Peter V o w d e n , David Wilkinson and Ralph C. Kester
Departments of Vascular Surgery and Neuroradiology*, St ]ames's University Hospital, Leeds, and Pinderfields Hospital, Wakefield, U.K.
Introduction A traumatic vertebral artery arteriovenous (A-V) fistula is a rare complication of a stab wound to the neck. Such lesions are difficult to treat and may have serious consequences for the patient. A direct surgical approach, which may require craniotomy and ligation of the involved vertebral artery, has been the treatment of choice. ~ A recently developed alternative is to employ an endovascular approach with balloon embolisation.2, 3 We report a case managed by embolisation and external compression manoeuvres and review the possible treatment options for vertebral A-V fistulae.
Case Report A 44-year-old man was referred to the Vascular Surgery Unit, St James's University Hospital, Leeds in February 1986 complaining of an annoying rushing noise in his left ear. Six months earlier he had been stabbed twice high in the left side of the neck. The first stab appeared superficial and entered laterally close to the mastoid tip. The second entered behind the angle of the jaw and penetrated through to his mouth to the left of the tongue.
Please address all correspondenceto: M. Nelson, Department ofNeuroradiology,Pinderfields Hospital, Wakefield, U,K. 0950-821X/88/020133 +07 $03.00/0 © 1988 Grune&Stratton Ltd
There was no excessive bleeding, nor did any large haematoma form. Examination failed to reveal any vascular or neurological injury. The wounds were not explored and both healed without complication after simple suturing. From the time of the trauma the patient was aware of a noise which he localised high in the left side of the neck. The noise was sometimes pulsatile and at other times constant. It became subjectively louder with time, was present throughout the day and interfered with his sleep. Its intensity was reduced when he turned his head to the right. No relevant clinical abnormality was found when he was re-examined and an audiogram was found to be normal. On examination, at representation some 6 months after the stabbing, the patient appeared well. There was a very loud bruit centred just behind the tip of the left mastoid process. This could be traced down the left anterior triangle and into the upper part of the posterior triangle. On the right a soft bruit was audible in the upper part of the posterior triangle. A thrill was palpable over the left occipital bone below the nuchal line. The remainder of the general and neurological examination was normal. Computed Tomography (CT) with intravenous contrast enhancement showed an increase in vascular structures in the leff-pre-vertebral space in the upper neck (Fig. 1). There was an associated increase in the epidural vascular space antero-laterally on the left between C2 and C4. There was no bony lesion. Selective carotid and vertebral angiograms showed a
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1 m m balloons used in this patient measure 1 x 5 mm uninflated and 5.3 × 1 6 m m inflated to the maximum recommended volume of 0.15 ml. Each is delivered via a 4.9F radiopaque polyethylene introducer catheter (ID 0.044 in.). Once the balloon is inflated with contrast to a volume sufficient to impact it within a blood vessel, further pressure on the syringe lubricates the contact areas of catheter and balloon with contrast medium, and allows the catheter to be detached by gentle traction. The balloon embolisation procedure was performed in the Department of Neuroradiology at Pinderflelds Hospital, Wakefield. The pattern of flow in the vertebral arteries indicated that left vetebral artery occlusion would be tolerated if a trapping procedure were necessary. Therefore it was not necessary to have the patient Fig. 1. Axial CT scan at the level of C2 shows prominent vessels in the awake for neurological testing during the procedure. We paraspinal musculature and widening of the epidural vascular space on chose to employ general anaesthesia to ensure the the left (arrow). patient's comfort and to minimise movement during balloon placement. A right transfemoral approach was used and a 5 F sheath inserted to minimise trauma to the high flow A-V fistula of the left vertebral artery at C1 artery. The patient was heparinised to reduce thrombolevel. This communicated with c e r v i c a l muscular and embolic complications using the regimen of Debrun et al. s epidural veins, the left internal jugular vein in the neck After preliminary check angiograms the 4.9 F introducer and the left cavernous sinus and superior ophthalmic catheter was inserted over a long exchange guide wire. vein intracranially (Fig. 2). There was angiographic evi- The tip was placed free of obstruction in the second segdence of intracranial steal and the left vertebral artery ment of the left vertebral artery, at mid-cervical level. Using Beckton-Dickinson 1 m m balloons the neck of emptied entirely into the fistula. The right vertebral artery filled only the lower half of the basilar artery and the fistula was found to be too wide for a stable lie to be there was intracranial reflux to the third part of the left achieved with occlusion of the fistula and sparing of the vertebral artery with substantial filling of the fistula, A vertebral artery. A safe detachment could not be made at further contribution to the fistula came from the second the fistula. Therapeutic options were either a trapping intervertebral space anastomotic branch of the left occipi- procedure with obstruction of the vertebral artery above tal artery but there was no internal carotid artery supply. and below the fistula or balloon occlusion of the veins The patient was discharged after the diagnostic study. draining the fistula. We chose the latter. A BecktonFor surgical treatment the high location of this Dickinson l mm balloon filled with 0 . 1 m l Iohexol lesion would have necessitated sub-occipital craniotomy, 220 mg/ml (Omnipaque, Nycomed Co.) was detached in Therefore we elected to treat this fistula by therapeutic veins close to the fistula. It obstructed the venous flow embolisation with a balloon occlusion technique as we posteriorly to the epidural and cervical muscular veins. considered that this carried less risk and potential mor- An interval arteriogram (not illustrated) showed inbidity. The patient was seen as an out-patient and the creased venous flow intracrania!ly to the left superior procedure of balloon embolisation and its potential risks ophthalmic vein. Since this presented an increased risk to ,were explained, Written consent was sought only after a the patient of visual problems, including the possibility of further interval which allowed the patient to discuss the t an ischaemic optic neuropathy, a second balloon was situation with his family. manipulated to a position where it obstructed the intraThere is a recent complete account of the balloon cranial venous outflow of the fistula. Immediate postcatheter system employed. 4 The Bard-Parker mini- embolisation angiograms showed that the high flow balloon detachable system (Beckton-Dickinson & Co., through the fistula had been considerably reduced (Fig. New Jersey, U.S.A.) is one of several balloon embolisation 3). Contrast reached the posterior cerebral arteries on left systems which are commercially available. The silicone vertebral angiograms and right vertebral angiograms balloons are provided mounted on a 150 cm long radi- showed normal filling of the basilar artery and its opaque polyurethane catheter (0.6 m m OD, 0.3 m m ID) branches with only minimal supply to the fistula. There with an intervening stainless steel, pressure-activated was no intracranial venous drainage. valve pin which renders the balloon detachable and self Immediately after the procedure the patient was no sealing, Two balloon sizes are available. The smaller longer aware of his bruit in normal circumstances Eur I VascSurg Vol 2, April 1988
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Fig. 2. Pre-embolisation angiograms: (a) Left vertebral lateral yiew and (b) Right vertebral AP view show the high-flow left vertebral A-V fistula with arterial supply from both vertebral arteries and venous drainage in several directions (see text). The basilar artery fills poorly and only from the right (arrow in (b)). (c) Left occipital lateral view shows a supply to the fistula from the second invertebral space anastomosis (arrow) via the vertebral artery. The appearance indicates a secondary supply to the fistula, not a second fistula.
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Fig. 3. Immediate post-embolisation angiograms: Left vertebral AP (a) and lateral (b) views and right vertebral AP (c) and lateral (d) views. Two balloons are in situ (arrows) and flow through the fistula is reduced. The basilar artery fills well and Venous drainage posterioily and intracranially has been eliminated. i
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~a) (b) Fig. 4. Fivemonths post-embolisationanglograms: Leftvertebral AP (a) and lateral (b) views. There is no residual flowthrough the fistula. A small pseudo-aneurysmis present at the site ofthe fistula.
though he coud hear it with concentratioh in a quiet room. The left occipital thrill was still palpable and was thought to be due to the occipita ! artery and its C1 collateral branch. To promote resolution of the residual low flow fistula the patient was taught two compression manoeuvres. By turning his hea.d sharply to the righ t and pressing on the left retromastoid region with his left hand it was hoped he would reduce blood flow through the left vertebral artery and through the C1 collateral branch of the left occipital artery. He was asked to maintain this posture for 10 rain in every waking hour. After 2 weeks the left occipital thrill seemed weaker and he found it more difficult to hear the bruit. By 3 weeks neither was apparent. There was no headache, neck stiffness nor any febrile illness •during this period: Follow-up arteriograms 5 months postembolisation showed occlusion of the fistula and a small residual pseudo-aneurysm on the C1 segment of the left vertebral artery (Fig. 4). The left vertebral artery now filled the basilar artery normally with some reflux down the right vertebral artery, The C1 collateral branch of the left occipital artery was no longer hypertrophic. The balloons remained inflated. Eighteen months after the embolisation no abnormality was apparent on clinical examination.
Discussion The clinical presentation and conventional surgical approaches to traumatic vertebral A-V fistulae have been reviewed recently by Miller et al.6 Endovascular occlusive technique s have various advantages over conventional surgical methods for the treatment of vertebral A-V fistulae. r Access to the fistula is simpler by a transvascular approach a n d does not require extensive dissection of the neck, mandibular splitting or suboccipital craniotomy. The site of the fistula is readily identified using rapid sequence angiography which separates the arterial and venous components of the lesion. Surgical identification of the fistula can be hindered by overlying arterialised veins or a pulsating false aneurysm. Since balloon embolisation techniques rely on the presence of flow towards the fistula it is not necessary to secure control of blood flow in the vertebral artery either proximal or distal to the fistula. A combination of local anaesthesia and mild sedation is usually sufficient for endovascular occlusion procedures. The problems with general anaesthesia are avoided and if it is necessary to trap the fistula the patient can be examined neurologically during a test occlusion of the vertebral artery before balloon detachment. The flow Eur I VascSurg Vol 2, April 1988
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pattern we observed in our patient allowed us to safely employ general anaesthesia. An unsuccessful attempt at endovascular occlusion does not preclude a later conventional surgical approach whereas unsuccessful surgery with vertebral artery ligation interferes with later endovascular treatment by impairing vascular access. Vertebral A-V fistulae may be treated by placing occlusive balloons at various sites in relation to the fistula. The balloon may be detached in the fistula to produce occlusion of the fistula and preservation of the vertebral artery. 2 Similar results can be achieved by detaching several balloons on the venous side of the fistula. a In this situation the balloons must occlude the venous outflow channels since simple packing of a false aneurysm is not sufficient to cause thrombosis. 6 Occlusion was achieved in 17 of 22 cases reported by Debrun et al. 2 using this method. In other situations the vertebral artery must be sacrificed. If the fistula is wide a large balloon inflated in the fistula may encroach on the vertebral artery and occlude it. When a balloon cannot be detached safely in the fistula a trapping procedure is necessary with occlusion of the vertebral artery. Balloons are placed in the vertebral artery proximal and distal to the fistula to exclude it from antegrade and retrograde vertebral supply. The balloons are placed close to the fistula to minimise the potential for the development of collateral supply from small muscular or spinal arteries. 6 Vascular access to vertebral A-V fistulae is usually obtained from the ipsilateral vertebral artery in an antegrade direction using a percutaneous transfemoral approach. Other approaches have been described. Goodm a n et al. 8 introduced two Fogarty balloon catheters through a proximal vertebral arteriotomy after surgical trapping with a distal clip and proximal ligation failed to abolish the shunt through a C2/3 fistula. Motet et al. 9 treated a congenital C1/2 fistula with a Fogarty balloon catheter which was passed through the anastomotic artery of the second cervical intervertebral space, from an occipital arteriotomy. They felt that this approach carried less risk of balloon migration in the vertebro-basilar territory. A traumatic C1 fistula was treated by Miller et al. 6 by trapping it proximally and distally with Hieshima detachable balloons. This could not be occluded directly because the vertebral artery was transected. The distal balloon was placed by catheterising the contralateral vertebral artery, passing it via the origin of the basilar artery, in a retrograde direction, to the distal segment of the vertebral artery on the affected side. Merland et al. 1 occluded a fistula with a Debrun balloon using a venous approach. In this case a direct approach through the ipsilateral vertebral artery was not possible because of proximal occlusion of the artery. Vertebral A-V fistulae may take an arterial supply from sources other than the vertebral artery, such as the Eur ] VascSurg Vol 2, April 1988
ipsilateral occipital or ascending cervical arteries. These associated supplies are of two haemodynamically distinct types. Less significant is the type illustrated by the occipital supply in our patient and in the report of Miller et al. 6 The shunt from these is abolished by occlusion of the vertebral fistula; they tend to be smaller and do not need to be embolised separately. More significant is the type fed by the occipital and ascending cervical arteries as in Case 1 in the report of Debrun et al.2 The shunt from these is not abolished by occlusion of the vertebral fistula. They represent separate A-V fistulae. They tend to be larger and must be embolised separately. Careful assessment of selective angiograms should enable the two types to be distinguished before embolisation is undertaken. When vertebral A-V fistulae are treated with balloon embolisation in venous structures distal to the fistula a residual false aneurysm at the site of the fistula is inevitable. 7 The natural history of these false aneurysms of the vertebral artery appears benign. The aneurysm in our patient became smaller in the 5 months after embolisation. In two cases the aneurysms have disappeared on follow-up angiograms done a few months and one year respectively after embolisation.l There is no report of a vertebral false aneurysm increasing in size nor of any aneurysm being associated with pain, nerve palsy or thrombo-embolic complications. This is remarkable considering the well recognised association of these problems with false aneurysms of the internal carotid artery after otherwise successful treatment of direct caroticocavernous fistulae. "~ External compression manoeuvres have been shown to be useful in the treatment of direct carotico-cavernous fistulae, /tagashida et al. 11 produced progressive and complete closure without recurrence at 12-months follow-up in eight of 48 patients (17%). They used a simple manoeuvre for simultaneous intermittent compression of the cervical carotid artery and jugular vein. Apart from one dramatic case reported by Matas 12 compression manoeuvres have not been applied in the treatment of vertebral A-V fistulae. Mechanical compression of the vertebral artery and the internal jugular vein have been demonstrated at various sites. These depend on the position of the head and neck, and on the presence of pathology in the osteoligamentous structures of the cervical spine. Ipsilateral rotation of the head compresses the pre-vertebral segment of the vertebral artery between the longus colli and scalenus anticus muscles. 1~ In children this also compresses the internal jugular vein at mid-cervical level, probably by stretching it against the sternomastoid muscle. 14 Contralateral rotation of the head may also compress the vertebral artery. This occurs after it enters the foramen transversarium~of C6 at the C 5 / 6 intervertebral foramen, at the atlanto-axial facet joint and at the
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atlanto-occipital joint, is Hyperextension of the neck, hypermobility at the atlanto-axial joints and osteophytes at the neurocentral joints m a y exaggerate these changes. '6 Intermittent vertebral artery occlusion has been described in a patient with rheumatoid arthritis. This occurred on ipsilateral head rotation or head extension and was t h o u g h t to be due to osteophytes a n d capsular laxity at the atlanto-axial facet joint.'7 The compression m a n o e u v r e applied by o u r patient was a m a x i m a l head rotation to the right, contralateral to the fistula, a n d direct pressure on the occipital artery thrill in the left retromastoid region using the t h e n a r eminence of his left hand. He was asked to do this for ten minutes in every waking h o u r and we would estimate that a compliance of about 80% was achieved. It is n o t certain that these measures were responsible for the abolition of the residual flow t h r o u g h the fistula. Spontaneous resolution of vertebral A-V fistulae has been d o c u m e n t e d in both u n t r e a t e d patients'S-~ and incompletely treated cases. 2,6 However, there was a close temporal relationship between the start of the compression m a n o e u v r e s and the loss of o u r patient's bruit. This leads us to postulate that such measures are w o r t h a trial in n o n - u r g e n t untreated cases or as an adjunct to endovascular or surgical treatment of vertebral artery A - V fistulae.
Acknowledgements We wish to thank Mr. M. M. Cameron, Consultant Neurosurgeon, Pinderfields Hospital, for his care of the patient whilst in Pinderfields Hospital and Dr. A. Shanks, Anaesthetist. We are also grateful for the assistance of the Department of Medical Illustration of the General Infirmary at Leeds.
References 1 MERLANDJ-J, REIZINED, RlCHE M-C et al. Endovascular treatment of vertebral arteriovenous fistulas in twenty-two patients. Ann Vasc Surg 1986 ; 1 : 73-78. 2 DEBRUN GM, LEGRE], KASBARIAN M, TAPIAS PL, CARONJP. Endovascular occlusion of vertebral fistulae by detachable balloons with conservation of the vertebral blood flow. Radiology 1979 ; 1 3 0 : 1 4 1 147.
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3 FA1RMAN R, GROSSMAN R, GONBERG H. A n e w approach to the treatment of vertebral arteriovenous fistulas. Surgery 1984;95 : l 12 114. 4 NORMAN D, NEWTON TH, El)WARDS MS, DECAPRIO V. Carotidcavernous fistula: Closure with detachable silicone balloons. Radiology 1 9 8 3 ; 1 4 9 : 1 4 9 - 1 5 7 . 5 DEBRUNGM, VINUELAFV, FOX Aj. Aspirin and systemic heparinization in diagnostic and interventional neuroradiotogy. Am ] Radiol ogy 1 9 8 2 ; 1 3 9 : 1 3 9 - 1 4 2 . 6 MILLER RE, HIESHIMA GB, GIANOTTA SL, GRINNEI.L VS, MEHRINGER CM, KERIN DS. Acute traumatic vertebral arteriovenous fistula; balloon occlusion with the use of a contralateral approach. Nemv surgery 1984; 14:225-229. 7 SCIALFA G, VAGtII A, VALSECCHI F, BERNARDI L, TONON (2. Neuroradiological treatment of carotid and vertebral fistulas and intracavernous aneurysms. Technical problems and results. Neuroradiology 1982 ; 24:13 25. 8 GOODMAN SJ, HASSO A, KIRKPATRICK D, Treatment of vertebrojugular fistula by balloon occlusion. ] Neurosurgery 1975 ; 43 : 362-367. 9 MORETJ, LASJAIJNIASF, DOYOND. Occipital approach for treatment of arteriovenous malformation of the vertebral artery by balloon occlusion. Neuroradiology 1979 ; 17: 269 273. 10 DEBRUN GM, LACOURP, VINUELA F, FOX A, DRAKE CG, CARON JP. Treatment of 54 traumatic carotid-cavernous fistulas. ] Nearosurgery 1981 ; 55 : 678-692. 11 ITIGASHIDART, HIESHIMA GB, HALBACH VV, BEATSONJR, GOTO K. Closure of carotid cavernous sinus fistulae by external compression of the carotid artery and jugular vein. Acta Radiologica 1986; [Suppl.] 369:580-583. 12 MATASR. Traumatisms and traumatic aneurisms of the vertebral artery and their surgical treatment, with the report of a cured case. AnnSurg 1 8 9 3 ; 1 8 : 4 7 7 - 5 2 1 . 13 Husm EA, BELL HS, STORERJ. Mechanical obstruction of the vertebral artery: A new clinical concept. JAMA 1966; 196:475-478. 14 WATSONGH. Effect of head rotation on jugular vein blood flow. Arch Dis Child 1 9 7 4 ; 4 9 : 2 3 7 - 2 3 9 . 15 SCHNEn)ERRC, SCHEMg GW. Vertebral artery insufficiency in acute and chronic spinal trauma, with special reference to the syndrome of acute central cervical spinal cord injury. J Neurosm'gery 1961; 18:348-360. 16 TATLOW WFT, BAMMER HG. Syndrome of vertebral compression, Neurology 1 9 5 7 ; 7 : 3 3 1 - 3 4 0 . 17 ROmNSON BP, SEEC,ER JF, ZAK SM. Rheumatoid arthritis and positional vertebrobasilar insufficiency. J Neurosurgery 1986;65: 111-114. ] 8 BERGOUISTE. Bilateral arteriovenous fistulae. A complication of vertebral angiography by direct percutaneous puncture. Br J Radiology 1971;44:519-523. 19 GOODDYW, SCHECHTERMM. Spontaneous arterio-venous fistula of the vertebral artery. Br ] Radiology 1960; 33 : 709-711. 20 NEWTONTH, DARROCHJ. Vertebral arteriovenous fistula complicating vertebral angiography. Acta Radiol (Diagn) ] 966; 5:428M40. 21 SHERMH, MEYERNL LENHARDTHF, TRUMMERMJ. Arteriovenous fistula involving the vertebral artery: Report of three cases. Ann Surg 1966; 163:408-413.
Received 14 September 1987
Eur] Vase Surg Vol 2, April 1988
CASE REPORT T r e a t m e n t of T r a u m a t i c Vertebral Arteriovenous Fistula by Balloon Occlusion and Compressive M a n o e u v r e s Michael Nelson*, Peter V o w d e n , David Wilkinson and Ralph C. Kester
Departments of Vascular Surgery and Neuroradiology*, St ]ames's University Hospital, Leeds, and Pinderfields Hospital, Wakefield, U.K.
Introduction A traumatic vertebral artery arteriovenous (A-V) fistula is a rare complication of a stab wound to the neck. Such lesions are difficult to treat and may have serious consequences for the patient. A direct surgical approach, which may require craniotomy and ligation of the involved vertebral artery, has been the treatment of choice. ~ A recently developed alternative is to employ an endovascular approach with balloon embolisation.2, 3 We report a case managed by embolisation and external compression manoeuvres and review the possible treatment options for vertebral A-V fistulae.
Case Report A 44-year-old man was referred to the Vascular Surgery Unit, St James's University Hospital, Leeds in February 1986 complaining of an annoying rushing noise in his left ear. Six months earlier he had been stabbed twice high in the left side of the neck. The first stab appeared superficial and entered laterally close to the mastoid tip. The second entered behind the angle of the jaw and penetrated through to his mouth to the left of the tongue.
Please address all correspondenceto: M. Nelson, Department ofNeuroradiology,Pinderfields Hospital, Wakefield, U,K. 0950-821X/88/020133 +07 $03.00/0 © 1988 Grune&Stratton Ltd
There was no excessive bleeding, nor did any large haematoma form. Examination failed to reveal any vascular or neurological injury. The wounds were not explored and both healed without complication after simple suturing. From the time of the trauma the patient was aware of a noise which he localised high in the left side of the neck. The noise was sometimes pulsatile and at other times constant. It became subjectively louder with time, was present throughout the day and interfered with his sleep. Its intensity was reduced when he turned his head to the right. No relevant clinical abnormality was found when he was re-examined and an audiogram was found to be normal. On examination, at representation some 6 months after the stabbing, the patient appeared well. There was a very loud bruit centred just behind the tip of the left mastoid process. This could be traced down the left anterior triangle and into the upper part of the posterior triangle. On the right a soft bruit was audible in the upper part of the posterior triangle. A thrill was palpable over the left occipital bone below the nuchal line. The remainder of the general and neurological examination was normal. Computed Tomography (CT) with intravenous contrast enhancement showed an increase in vascular structures in the leff-pre-vertebral space in the upper neck (Fig. 1). There was an associated increase in the epidural vascular space antero-laterally on the left between C2 and C4. There was no bony lesion. Selective carotid and vertebral angiograms showed a
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1 m m balloons used in this patient measure 1 x 5 mm uninflated and 5.3 × 1 6 m m inflated to the maximum recommended volume of 0.15 ml. Each is delivered via a 4.9F radiopaque polyethylene introducer catheter (ID 0.044 in.). Once the balloon is inflated with contrast to a volume sufficient to impact it within a blood vessel, further pressure on the syringe lubricates the contact areas of catheter and balloon with contrast medium, and allows the catheter to be detached by gentle traction. The balloon embolisation procedure was performed in the Department of Neuroradiology at Pinderflelds Hospital, Wakefield. The pattern of flow in the vertebral arteries indicated that left vetebral artery occlusion would be tolerated if a trapping procedure were necessary. Therefore it was not necessary to have the patient Fig. 1. Axial CT scan at the level of C2 shows prominent vessels in the awake for neurological testing during the procedure. We paraspinal musculature and widening of the epidural vascular space on chose to employ general anaesthesia to ensure the the left (arrow). patient's comfort and to minimise movement during balloon placement. A right transfemoral approach was used and a 5 F sheath inserted to minimise trauma to the high flow A-V fistula of the left vertebral artery at C1 artery. The patient was heparinised to reduce thrombolevel. This communicated with c e r v i c a l muscular and embolic complications using the regimen of Debrun et al. s epidural veins, the left internal jugular vein in the neck After preliminary check angiograms the 4.9 F introducer and the left cavernous sinus and superior ophthalmic catheter was inserted over a long exchange guide wire. vein intracranially (Fig. 2). There was angiographic evi- The tip was placed free of obstruction in the second segdence of intracranial steal and the left vertebral artery ment of the left vertebral artery, at mid-cervical level. Using Beckton-Dickinson 1 m m balloons the neck of emptied entirely into the fistula. The right vertebral artery filled only the lower half of the basilar artery and the fistula was found to be too wide for a stable lie to be there was intracranial reflux to the third part of the left achieved with occlusion of the fistula and sparing of the vertebral artery with substantial filling of the fistula, A vertebral artery. A safe detachment could not be made at further contribution to the fistula came from the second the fistula. Therapeutic options were either a trapping intervertebral space anastomotic branch of the left occipi- procedure with obstruction of the vertebral artery above tal artery but there was no internal carotid artery supply. and below the fistula or balloon occlusion of the veins The patient was discharged after the diagnostic study. draining the fistula. We chose the latter. A BecktonFor surgical treatment the high location of this Dickinson l mm balloon filled with 0 . 1 m l Iohexol lesion would have necessitated sub-occipital craniotomy, 220 mg/ml (Omnipaque, Nycomed Co.) was detached in Therefore we elected to treat this fistula by therapeutic veins close to the fistula. It obstructed the venous flow embolisation with a balloon occlusion technique as we posteriorly to the epidural and cervical muscular veins. considered that this carried less risk and potential mor- An interval arteriogram (not illustrated) showed inbidity. The patient was seen as an out-patient and the creased venous flow intracrania!ly to the left superior procedure of balloon embolisation and its potential risks ophthalmic vein. Since this presented an increased risk to ,were explained, Written consent was sought only after a the patient of visual problems, including the possibility of further interval which allowed the patient to discuss the t an ischaemic optic neuropathy, a second balloon was situation with his family. manipulated to a position where it obstructed the intraThere is a recent complete account of the balloon cranial venous outflow of the fistula. Immediate postcatheter system employed. 4 The Bard-Parker mini- embolisation angiograms showed that the high flow balloon detachable system (Beckton-Dickinson & Co., through the fistula had been considerably reduced (Fig. New Jersey, U.S.A.) is one of several balloon embolisation 3). Contrast reached the posterior cerebral arteries on left systems which are commercially available. The silicone vertebral angiograms and right vertebral angiograms balloons are provided mounted on a 150 cm long radi- showed normal filling of the basilar artery and its opaque polyurethane catheter (0.6 m m OD, 0.3 m m ID) branches with only minimal supply to the fistula. There with an intervening stainless steel, pressure-activated was no intracranial venous drainage. valve pin which renders the balloon detachable and self Immediately after the procedure the patient was no sealing, Two balloon sizes are available. The smaller longer aware of his bruit in normal circumstances Eur I VascSurg Vol 2, April 1988
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Fig. 2. Pre-embolisation angiograms: (a) Left vertebral lateral yiew and (b) Right vertebral AP view show the high-flow left vertebral A-V fistula with arterial supply from both vertebral arteries and venous drainage in several directions (see text). The basilar artery fills poorly and only from the right (arrow in (b)). (c) Left occipital lateral view shows a supply to the fistula from the second invertebral space anastomosis (arrow) via the vertebral artery. The appearance indicates a secondary supply to the fistula, not a second fistula.
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Fig. 3. Immediate post-embolisation angiograms: Left vertebral AP (a) and lateral (b) views and right vertebral AP (c) and lateral (d) views. Two balloons are in situ (arrows) and flow through the fistula is reduced. The basilar artery fills well and Venous drainage posterioily and intracranially has been eliminated. i
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~a) (b) Fig. 4. Fivemonths post-embolisationanglograms: Leftvertebral AP (a) and lateral (b) views. There is no residual flowthrough the fistula. A small pseudo-aneurysmis present at the site ofthe fistula.
though he coud hear it with concentratioh in a quiet room. The left occipital thrill was still palpable and was thought to be due to the occipita ! artery and its C1 collateral branch. To promote resolution of the residual low flow fistula the patient was taught two compression manoeuvres. By turning his hea.d sharply to the righ t and pressing on the left retromastoid region with his left hand it was hoped he would reduce blood flow through the left vertebral artery and through the C1 collateral branch of the left occipital artery. He was asked to maintain this posture for 10 rain in every waking hour. After 2 weeks the left occipital thrill seemed weaker and he found it more difficult to hear the bruit. By 3 weeks neither was apparent. There was no headache, neck stiffness nor any febrile illness •during this period: Follow-up arteriograms 5 months postembolisation showed occlusion of the fistula and a small residual pseudo-aneurysm on the C1 segment of the left vertebral artery (Fig. 4). The left vertebral artery now filled the basilar artery normally with some reflux down the right vertebral artery, The C1 collateral branch of the left occipital artery was no longer hypertrophic. The balloons remained inflated. Eighteen months after the embolisation no abnormality was apparent on clinical examination.
Discussion The clinical presentation and conventional surgical approaches to traumatic vertebral A-V fistulae have been reviewed recently by Miller et al.6 Endovascular occlusive technique s have various advantages over conventional surgical methods for the treatment of vertebral A-V fistulae. r Access to the fistula is simpler by a transvascular approach a n d does not require extensive dissection of the neck, mandibular splitting or suboccipital craniotomy. The site of the fistula is readily identified using rapid sequence angiography which separates the arterial and venous components of the lesion. Surgical identification of the fistula can be hindered by overlying arterialised veins or a pulsating false aneurysm. Since balloon embolisation techniques rely on the presence of flow towards the fistula it is not necessary to secure control of blood flow in the vertebral artery either proximal or distal to the fistula. A combination of local anaesthesia and mild sedation is usually sufficient for endovascular occlusion procedures. The problems with general anaesthesia are avoided and if it is necessary to trap the fistula the patient can be examined neurologically during a test occlusion of the vertebral artery before balloon detachment. The flow Eur I VascSurg Vol 2, April 1988
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pattern we observed in our patient allowed us to safely employ general anaesthesia. An unsuccessful attempt at endovascular occlusion does not preclude a later conventional surgical approach whereas unsuccessful surgery with vertebral artery ligation interferes with later endovascular treatment by impairing vascular access. Vertebral A-V fistulae may be treated by placing occlusive balloons at various sites in relation to the fistula. The balloon may be detached in the fistula to produce occlusion of the fistula and preservation of the vertebral artery. 2 Similar results can be achieved by detaching several balloons on the venous side of the fistula. a In this situation the balloons must occlude the venous outflow channels since simple packing of a false aneurysm is not sufficient to cause thrombosis. 6 Occlusion was achieved in 17 of 22 cases reported by Debrun et al. 2 using this method. In other situations the vertebral artery must be sacrificed. If the fistula is wide a large balloon inflated in the fistula may encroach on the vertebral artery and occlude it. When a balloon cannot be detached safely in the fistula a trapping procedure is necessary with occlusion of the vertebral artery. Balloons are placed in the vertebral artery proximal and distal to the fistula to exclude it from antegrade and retrograde vertebral supply. The balloons are placed close to the fistula to minimise the potential for the development of collateral supply from small muscular or spinal arteries. 6 Vascular access to vertebral A-V fistulae is usually obtained from the ipsilateral vertebral artery in an antegrade direction using a percutaneous transfemoral approach. Other approaches have been described. Goodm a n et al. 8 introduced two Fogarty balloon catheters through a proximal vertebral arteriotomy after surgical trapping with a distal clip and proximal ligation failed to abolish the shunt through a C2/3 fistula. Motet et al. 9 treated a congenital C1/2 fistula with a Fogarty balloon catheter which was passed through the anastomotic artery of the second cervical intervertebral space, from an occipital arteriotomy. They felt that this approach carried less risk of balloon migration in the vertebro-basilar territory. A traumatic C1 fistula was treated by Miller et al. 6 by trapping it proximally and distally with Hieshima detachable balloons. This could not be occluded directly because the vertebral artery was transected. The distal balloon was placed by catheterising the contralateral vertebral artery, passing it via the origin of the basilar artery, in a retrograde direction, to the distal segment of the vertebral artery on the affected side. Merland et al. 1 occluded a fistula with a Debrun balloon using a venous approach. In this case a direct approach through the ipsilateral vertebral artery was not possible because of proximal occlusion of the artery. Vertebral A-V fistulae may take an arterial supply from sources other than the vertebral artery, such as the Eur ] VascSurg Vol 2, April 1988
ipsilateral occipital or ascending cervical arteries. These associated supplies are of two haemodynamically distinct types. Less significant is the type illustrated by the occipital supply in our patient and in the report of Miller et al. 6 The shunt from these is abolished by occlusion of the vertebral fistula; they tend to be smaller and do not need to be embolised separately. More significant is the type fed by the occipital and ascending cervical arteries as in Case 1 in the report of Debrun et al.2 The shunt from these is not abolished by occlusion of the vertebral fistula. They represent separate A-V fistulae. They tend to be larger and must be embolised separately. Careful assessment of selective angiograms should enable the two types to be distinguished before embolisation is undertaken. When vertebral A-V fistulae are treated with balloon embolisation in venous structures distal to the fistula a residual false aneurysm at the site of the fistula is inevitable. 7 The natural history of these false aneurysms of the vertebral artery appears benign. The aneurysm in our patient became smaller in the 5 months after embolisation. In two cases the aneurysms have disappeared on follow-up angiograms done a few months and one year respectively after embolisation.l There is no report of a vertebral false aneurysm increasing in size nor of any aneurysm being associated with pain, nerve palsy or thrombo-embolic complications. This is remarkable considering the well recognised association of these problems with false aneurysms of the internal carotid artery after otherwise successful treatment of direct caroticocavernous fistulae. "~ External compression manoeuvres have been shown to be useful in the treatment of direct carotico-cavernous fistulae, /tagashida et al. 11 produced progressive and complete closure without recurrence at 12-months follow-up in eight of 48 patients (17%). They used a simple manoeuvre for simultaneous intermittent compression of the cervical carotid artery and jugular vein. Apart from one dramatic case reported by Matas 12 compression manoeuvres have not been applied in the treatment of vertebral A-V fistulae. Mechanical compression of the vertebral artery and the internal jugular vein have been demonstrated at various sites. These depend on the position of the head and neck, and on the presence of pathology in the osteoligamentous structures of the cervical spine. Ipsilateral rotation of the head compresses the pre-vertebral segment of the vertebral artery between the longus colli and scalenus anticus muscles. 1~ In children this also compresses the internal jugular vein at mid-cervical level, probably by stretching it against the sternomastoid muscle. 14 Contralateral rotation of the head may also compress the vertebral artery. This occurs after it enters the foramen transversarium~of C6 at the C 5 / 6 intervertebral foramen, at the atlanto-axial facet joint and at the
T r e a t m e n t of V e r t e b r a l A r t e r i o v e n o u s F i s t u l a e
atlanto-occipital joint, is Hyperextension of the neck, hypermobility at the atlanto-axial joints and osteophytes at the neurocentral joints m a y exaggerate these changes. '6 Intermittent vertebral artery occlusion has been described in a patient with rheumatoid arthritis. This occurred on ipsilateral head rotation or head extension and was t h o u g h t to be due to osteophytes a n d capsular laxity at the atlanto-axial facet joint.'7 The compression m a n o e u v r e applied by o u r patient was a m a x i m a l head rotation to the right, contralateral to the fistula, a n d direct pressure on the occipital artery thrill in the left retromastoid region using the t h e n a r eminence of his left hand. He was asked to do this for ten minutes in every waking h o u r and we would estimate that a compliance of about 80% was achieved. It is n o t certain that these measures were responsible for the abolition of the residual flow t h r o u g h the fistula. Spontaneous resolution of vertebral A-V fistulae has been d o c u m e n t e d in both u n t r e a t e d patients'S-~ and incompletely treated cases. 2,6 However, there was a close temporal relationship between the start of the compression m a n o e u v r e s and the loss of o u r patient's bruit. This leads us to postulate that such measures are w o r t h a trial in n o n - u r g e n t untreated cases or as an adjunct to endovascular or surgical treatment of vertebral artery A - V fistulae.
Acknowledgements We wish to thank Mr. M. M. Cameron, Consultant Neurosurgeon, Pinderfields Hospital, for his care of the patient whilst in Pinderfields Hospital and Dr. A. Shanks, Anaesthetist. We are also grateful for the assistance of the Department of Medical Illustration of the General Infirmary at Leeds.
References 1 MERLANDJ-J, REIZINED, RlCHE M-C et al. Endovascular treatment of vertebral arteriovenous fistulas in twenty-two patients. Ann Vasc Surg 1986 ; 1 : 73-78. 2 DEBRUN GM, LEGRE], KASBARIAN M, TAPIAS PL, CARONJP. Endovascular occlusion of vertebral fistulae by detachable balloons with conservation of the vertebral blood flow. Radiology 1979 ; 1 3 0 : 1 4 1 147.
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3 FA1RMAN R, GROSSMAN R, GONBERG H. A n e w approach to the treatment of vertebral arteriovenous fistulas. Surgery 1984;95 : l 12 114. 4 NORMAN D, NEWTON TH, El)WARDS MS, DECAPRIO V. Carotidcavernous fistula: Closure with detachable silicone balloons. Radiology 1 9 8 3 ; 1 4 9 : 1 4 9 - 1 5 7 . 5 DEBRUNGM, VINUELAFV, FOX Aj. Aspirin and systemic heparinization in diagnostic and interventional neuroradiotogy. Am ] Radiol ogy 1 9 8 2 ; 1 3 9 : 1 3 9 - 1 4 2 . 6 MILLER RE, HIESHIMA GB, GIANOTTA SL, GRINNEI.L VS, MEHRINGER CM, KERIN DS. Acute traumatic vertebral arteriovenous fistula; balloon occlusion with the use of a contralateral approach. Nemv surgery 1984; 14:225-229. 7 SCIALFA G, VAGtII A, VALSECCHI F, BERNARDI L, TONON (2. Neuroradiological treatment of carotid and vertebral fistulas and intracavernous aneurysms. Technical problems and results. Neuroradiology 1982 ; 24:13 25. 8 GOODMAN SJ, HASSO A, KIRKPATRICK D, Treatment of vertebrojugular fistula by balloon occlusion. ] Neurosurgery 1975 ; 43 : 362-367. 9 MORETJ, LASJAIJNIASF, DOYOND. Occipital approach for treatment of arteriovenous malformation of the vertebral artery by balloon occlusion. Neuroradiology 1979 ; 17: 269 273. 10 DEBRUN GM, LACOURP, VINUELA F, FOX A, DRAKE CG, CARON JP. Treatment of 54 traumatic carotid-cavernous fistulas. ] Nearosurgery 1981 ; 55 : 678-692. 11 ITIGASHIDART, HIESHIMA GB, HALBACH VV, BEATSONJR, GOTO K. Closure of carotid cavernous sinus fistulae by external compression of the carotid artery and jugular vein. Acta Radiologica 1986; [Suppl.] 369:580-583. 12 MATASR. Traumatisms and traumatic aneurisms of the vertebral artery and their surgical treatment, with the report of a cured case. AnnSurg 1 8 9 3 ; 1 8 : 4 7 7 - 5 2 1 . 13 Husm EA, BELL HS, STORERJ. Mechanical obstruction of the vertebral artery: A new clinical concept. JAMA 1966; 196:475-478. 14 WATSONGH. Effect of head rotation on jugular vein blood flow. Arch Dis Child 1 9 7 4 ; 4 9 : 2 3 7 - 2 3 9 . 15 SCHNEn)ERRC, SCHEMg GW. Vertebral artery insufficiency in acute and chronic spinal trauma, with special reference to the syndrome of acute central cervical spinal cord injury. J Neurosm'gery 1961; 18:348-360. 16 TATLOW WFT, BAMMER HG. Syndrome of vertebral compression, Neurology 1 9 5 7 ; 7 : 3 3 1 - 3 4 0 . 17 ROmNSON BP, SEEC,ER JF, ZAK SM. Rheumatoid arthritis and positional vertebrobasilar insufficiency. J Neurosurgery 1986;65: 111-114. ] 8 BERGOUISTE. Bilateral arteriovenous fistulae. A complication of vertebral angiography by direct percutaneous puncture. Br J Radiology 1971;44:519-523. 19 GOODDYW, SCHECHTERMM. Spontaneous arterio-venous fistula of the vertebral artery. Br ] Radiology 1960; 33 : 709-711. 20 NEWTONTH, DARROCHJ. Vertebral arteriovenous fistula complicating vertebral angiography. Acta Radiol (Diagn) ] 966; 5:428M40. 21 SHERMH, MEYERNL LENHARDTHF, TRUMMERMJ. Arteriovenous fistula involving the vertebral artery: Report of three cases. Ann Surg 1966; 163:408-413.
Received 14 September 1987
Eur] Vase Surg Vol 2, April 1988