Brandt, T.

Herrmann, K.

Mayer, T. E.

Strupp, M.

Zingler, V. C.

English

Universität München: Elektronischen Publikationen

Short Reports
Published online: December 2, 2004
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Eur Neurol 2004;52:250–251
DOI: 10.1159/000082370
Stent Grafting Resolved Brachial Plexus
Neuropathy due to Cervical Arteriovenous
Fistula
Vera Carina Zingler a, Michael Strupp a, Thomas Brandt a,
Karin Herrmannb, Thomas E. Mayer c
Departments of a Neurology, b Radiology and
cNeuroradiology, University of Munich, Munich, Germany
We report a rare case of an iatrogenic vertebrovenous fistula pre-
senting with a brachial plexus neuropathy and a cervical bruit. This
arteriovenous fistula (AVF) was successfully occluded by implanting
a stent graft into the right vertebral artery. After the stenting, the
cervical bruit ceased and the radiculopathy slowly improved. This
case highlights the usefulness of such new devices as covered stent
grafts, and the therapeutic options for effectively treating AVFs while
maintaining patency of the vertebral artery.
Case Report
Spinal AVFs are characterized by the presenting symptoms of tin-
nitus, vertebrobasilar ischemia [1, 2], myelopathy or subarachnoid
hemorrhage [3]. Only a few cases of cervical AVF associated with
radiculopathy have been reported so far, because AVF rarely occurs
at this site [4–7]. We describe a case of progressive brachial neuropa-
thy that was caused by a vertebrovenous AVF. The patient was suc-
cessfully treated by stent grafting of the vertebral artery.
A 63-year-old woman was admitted to our hospital with a 3-
month history of progressive pain and fluctuating paresthesia of the
right arm. The patient history revealed that right-sided breast carci-
noma had been treated by surgery and local radiotherapy 4 years pre-
viously. Five months before onset of the brachial neuropathy, she
had undergone a liver transplantation for fulminant hepatitis A and
had received several central venous lines on the right side.
The neurological examination revealed hypesthesia and paresthe-
sia of the right upper limb with predominant involvement of the der-
matomes C6–C8. The muscle strength as well as the reflexes of both
upper and lower limbs were normal. A loud, continuous bruit was
detected during auscultation of the right lateral neck. Electromyogra-
phy showed that the amplitude of the compound action potential of
the right median nerve was reduced. Moreover, there were signs of
chronic denervation of the right interosseus muscle. Motor and sen-
sory electroneurography were normal. A recording of the somatosen-
sory-evoked potentials after stimulation of the right median nerve
revealed a loss of the peripheral responses (N13/14). Doppler ultra-
sound showed a pathological flow in the vertebral artery on the right
side. MRI of the cervical spine demonstrated ectatic veins with flow
Fig. 1. T2-weighted axial MRI at the level of C6/C7. Flow voids of
dilated veins (arrows) around the nerve routes on the right side.
voids in the neuroforamina on the right (fig. 1). Conventional angiog-
raphy disclosed a direct vertebral artery fistula draining into the
paravertebral venous plexus at the level of the root C7 (fig. 2a).
The AVF was occluded by implanting a Jomed stent graft (4 mm
! 1.2 cm) into the right vertebral artery (by T.E.M.). The stent was
dilated to 6 mm proximally with a Maverick balloon (fig. 2b). No
neurological complications occurred during or after the endovascular
treatment. The cervical bruit ceased, and the brachialgia and pares-
thesia of the right arm slowly improved after the stenting.
We had first thought that the right-sided brachial plexus syn-
drome was either caused by direct metastatic spread or was a delayed
complication of the prior local radiation therapy [8, 9]. However, the
cervical bruit pointed to an AVF that angiography confirmed. This
case of AVF was most likely iatrogenic, having occurred after an
unintentional puncture of the vertebral artery during insertion of a
central venous line. Most cervical AVFs are caused by trauma, but
they can also occur spontaneously or in association with other vascu-
lar dysplasias [5]. The brachial neuropathy of our patient was proba-
bly due to a direct compression of the cervical roots by dilated drain-
ing veins; this was documented by MRI and angiography (fig. 1, 2a).
Short Reports 251
Fig. 2. a Angiography of the right vertebral artery. Ectatic proximal vertebral artery, pseudoaneurysm and direct fistula (arrow)
with early filling of the paravertebral venous plexus. b Remodeling of the vertebral artery and occlusion of the AVF by the
polytetrafluoroethylene-covered stent graft (arrows).
Whereas in the past the most common treatment of AVF consisted of
embolization with microcoils or surgery, nowadays successful stent
repair has been reported in a few cases [10–12]. In our patient, a
coronary polytetrafluoroethylene-covered stent graft repair was suffi-
cient. It also successfully maintained patency of the vertebral artery
(fig. 2b). This procedure appears superior to surgery or embolization,
since patency of the vertebral artery is maintained.
References
1 Vinchon M, Laurian C, George B, et al: Vertebral arteriovenous fistula: A
study of 49 cases and review of the literature. Cardiovasc Surg 1994;2:359–
369.
2 Waitzman A, Anderson J, Willinsky R: Endovascular management of ver-
tebral arteriovenous fistulas: The Toronto experience. J Otolaryngol
1996;25:322–328.
3 Willinsky R, Ter Brugge K, Lasjaunias P, et al: The variable presentations
of craniocervical and cervical dural arteriovenous malformations. Surg
Neurol 1990;34:118–123.
4 Kohno M, Takahashi H, Ide K, et al: A cervical dural arteriovenous fistula
in a patient presenting with radiculopathy. J Neurosurg 1996;84:119–123.
5 Taylor CG, Husami Y, Colquhoun IR, Byrne JV: Direct cervical vertebro-
venous fistula with radiculopathy and MRI changes resolving after success-
ful endovascular embolisation: A report of two cases. Neuroradiology
2001;43:1118–1122.
6 Cahan LD, Higashida RT, Halbach VV, et al: Variants of radiculomenin-
geal vascular malformations of the spine. J Neurosurg 1987;66:333–337.
7 Beaujeux R, Reizine D, Casasco A, et al: Endovascular treatment of verte-
bral arteriovenous fistula. Radiology 1992;183:361–367.
8 Rubin DI, Schomberg PJ, Shepherd RF, Panneton JM: Arteritis and bra-
chial plexus neuropathy as delayed complications of radiation therapy.
Mayo Clin Proc 2001;76:849–852.
9 Fardin P, Lelli S, Negrin P, Maluta S: Radiation-induced brachial plexopa-
thy: Clinical and electromyographical (EMG) considerations in 13 cases.
Electromyogr Clin Neurophysiol 1990;30:277–282.
10 Priestley R, Bray P, Bray A, Hunter J: Iatrogenic vertebral arteriovenous
fistula treated with a hemobahn stent-graft. J Endovasc Ther 2003;10:657–
663.
11 Surber R, Werner GS, Cohnert TU, Wahlers T, Figulla HR: Recurrent ver-
tebral arteriovenous fistula after surgical repair: Treatment with a self-
expanding stent-graft. J Endovasc Ther 2003;10:49–53.
12 Gonzalez A, Mayol A, Gil-Peralta A, Gonzalez-Marcos JR: Endovascular
stent-graft treatment of an iatrogenic vertebral arteriovenous fistula. Neu-
roradiology 2001;43:784–786.
Vera Zingler, MD
Department of Neurology, University of Munich, Klinikum Grosshadern
Marchioninistrasse 15, DE–81377 Munich (Germany)
Tel. +49 89 7095 2585, Fax +49 89 7095 5584
E-Mail vzingler@nro.med.uni-muenchen.de


Stent grafting resolved brachial plexus neuropathy due to cervical arteriovenous fistula

Open Access LMU ( Ludwig-Maximilians-Univ. München)

Short ReportsPublished online: December 2, 2004ABCFax + 41 61 306 12 34E-Mail karger@karger.chwww.karger.com© 2004 S. Karger AG, BaselAccessible online at:www.karger.com/eneEur Neurol 2004;52:250–251DOI: 10.1159/000082370Stent Grafting Resolved Brachial PlexusNeuropathy due to Cervical ArteriovenousFistulaVera Carina Zingler a, Michael Strupp a, Thomas Brandt a,Karin Herrmannb, Thomas E. Mayer cDepartments of a Neurology, b Radiology andcNeuroradiology, University of Munich, Munich, GermanyWe report a rare case of an iatrogenic vertebrovenous fistula pre-senting with a brachial plexus neuropathy and a cervical bruit. Thisarteriovenous fistula (AVF) was successfully occluded by implantinga stent graft into the right vertebral artery. After the stenting, thecervical bruit ceased and the radiculopathy slowly improved. Thiscase highlights the usefulness of such new devices as covered stentgrafts, and the therapeutic options for effectively treating AVFs whilemaintaining patency of the vertebral artery.Case ReportSpinal AVFs are characterized by the presenting symptoms of tin-nitus, vertebrobasilar ischemia [1, 2], myelopathy or subarachnoidhemorrhage [3]. Only a few cases of cervical AVF associated withradiculopathy have been reported so far, because AVF rarely occursat this site [4–7]. We describe a case of progressive brachial neuropa-thy that was caused by a vertebrovenous AVF. The patient was suc-cessfully treated by stent grafting of the vertebral artery.A 63-year-old woman was admitted to our hospital with a 3-month history of progressive pain and fluctuating paresthesia of theright arm. The patient history revealed that right-sided breast carci-noma had been treated by surgery and local radiotherapy 4 years pre-viously. Five months before onset of the brachial neuropathy, shehad undergone a liver transplantation for fulminant hepatitis A andhad received several central venous lines on the right side.The neurological examination revealed hypesthesia and paresthe-sia of the right upper limb with predominant involvement of the der-matomes C6–C8. The muscle strength as well as the reflexes of bothupper and lower limbs were normal. A loud, continuous bruit wasdetected during auscultation of the right lateral neck. Electromyogra-phy showed that the amplitude of the compound action potential ofthe right median nerve was reduced. Moreover, there were signs ofchronic denervation of the right interosseus muscle. Motor and sen-sory electroneurography were normal. A recording of the somatosen-sory-evoked potentials after stimulation of the right median nerverevealed a loss of the peripheral responses (N13/14). Doppler ultra-sound showed a pathological flow in the vertebral artery on the rightside. MRI of the cervical spine demonstrated ectatic veins with flowFig. 1. T2-weighted axial MRI at the level of C6/C7. Flow voids ofdilated veins (arrows) around the nerve routes on the right side.voids in the neuroforamina on the right (fig. 1). Conventional angiog-raphy disclosed a direct vertebral artery fistula draining into theparavertebral venous plexus at the level of the root C7 (fig. 2a).The AVF was occluded by implanting a Jomed stent graft (4 mm! 1.2 cm) into the right vertebral artery (by T.E.M.). The stent wasdilated to 6 mm proximally with a Maverick balloon (fig. 2b). Noneurological complications occurred during or after the endovasculartreatment. The cervical bruit ceased, and the brachialgia and pares-thesia of the right arm slowly improved after the stenting.We had first thought that the right-sided brachial plexus syn-drome was either caused by direct metastatic spread or was a delayedcomplication of the prior local radiation therapy [8, 9]. However, thecervical bruit pointed to an AVF that angiography confirmed. Thiscase of AVF was most likely iatrogenic, having occurred after anunintentional puncture of the vertebral artery during insertion of acentral venous line. Most cervical AVFs are caused by trauma, butthey can also occur spontaneously or in association with other vascu-lar dysplasias [5]. The brachial neuropathy of our patient was proba-bly due to a direct compression of the cervical roots by dilated drain-ing veins; this was documented by MRI and angiography (fig. 1, 2a).Short Reports 251Fig. 2. a Angiography of the right vertebral artery. Ectatic proximal vertebral artery, pseudoaneurysm and direct fistula (arrow)with early filling of the paravertebral venous plexus. b Remodeling of the vertebral artery and occlusion of the AVF by thepolytetrafluoroethylene-covered stent graft (arrows).Whereas in the past the most common treatment of AVF consisted ofembolization with microcoils or surgery, nowadays successful stentrepair has been reported in a few cases [10–12]. In our patient, acoronary polytetrafluoroethylene-covered stent graft repair was suffi-cient. It also successfully maintained patency of the vertebral artery(fig. 2b). This procedure appears superior to surgery or embolization,since patency of the vertebral artery is maintained.References1 Vinchon M, Laurian C, George B, et al: Vertebral arteriovenous fistula: Astudy of 49 cases and review of the literature. Cardiovasc Surg 1994;2:359–369.2 Waitzman A, Anderson J, Willinsky R: Endovascular management of ver-tebral arteriovenous fistulas: The Toronto experience. J Otolaryngol1996;25:322–328.3 Willinsky R, Ter Brugge K, Lasjaunias P, et al: The variable presentationsof craniocervical and cervical dural arteriovenous malformations. SurgNeurol 1990;34:118–123.4 Kohno M, Takahashi H, Ide K, et al: A cervical dural arteriovenous fistulain a patient presenting with radiculopathy. J Neurosurg 1996;84:119–123.5 Taylor CG, Husami Y, Colquhoun IR, Byrne JV: Direct cervical vertebro-venous fistula with radiculopathy and MRI changes resolving after success-ful endovascular embolisation: A report of two cases. Neuroradiology2001;43:1118–1122.6 Cahan LD, Higashida RT, Halbach VV, et al: Variants of radiculomenin-geal vascular malformations of the spine. J Neurosurg 1987;66:333–337.7 Beaujeux R, Reizine D, Casasco A, et al: Endovascular treatment of verte-bral arteriovenous fistula. Radiology 1992;183:361–367.8 Rubin DI, Schomberg PJ, Shepherd RF, Panneton JM: Arteritis and bra-chial plexus neuropathy as delayed complications of radiation therapy.Mayo Clin Proc 2001;76:849–852.9 Fardin P, Lelli S, Negrin P, Maluta S: Radiation-induced brachial plexopa-thy: Clinical and electromyographical (EMG) considerations in 13 cases.Electromyogr Clin Neurophysiol 1990;30:277–282.10 Priestley R, Bray P, Bray A, Hunter J: Iatrogenic vertebral arteriovenousfistula treated with a hemobahn stent-graft. J Endovasc Ther 2003;10:657–663.11 Surber R, Werner GS, Cohnert TU, Wahlers T, Figulla HR: Recurrent ver-tebral arteriovenous fistula after surgical repair: Treatment with a self-expanding stent-graft. J Endovasc Ther 2003;10:49–53.12 Gonzalez A, Mayol A, Gil-Peralta A, Gonzalez-Marcos JR: Endovascularstent-graft treatment of an iatrogenic vertebral arteriovenous fistula. Neu-roradiology 2001;43:784–786.Vera Zingler, MDDepartment of Neurology, University of Munich, Klinikum GrosshadernMarchioninistrasse 15, DE–81377 Munich (Germany)Tel. +49 89 7095 2585, Fax +49 89 7095 5584E-Mail vzingler@nro.med.uni-muenchen.de

al: A cervical dural arteriovenous fistula in a patient presenting with radiculopathy.

al: The variable presentations of craniocervical and cervical dural arteriovenous malformations. Surg Neurol

Arteritis and brachial plexus neuropathy as delayed complications of radiation therapy. Mayo Clin Proc

Endovascular management of vertebral arteriovenous fistulas: The Toronto experience.

et al: Endovascular treatment of vertebral arteriovenous fistula. Radiology

et al: Variants of radiculomeningeal vascular malformations of the spine.

et al: Vertebral arteriovenous fistula: A study of 49 cases and review of the literature. Cardiovasc Surg

Figulla HR: Recurrent vertebral arteriovenous fistula after surgical repair: Treatment with a selfexpanding stent-graft.

Gonzalez-Marcos JR: Endovascular stent-graft treatment of an iatrogenic vertebral arteriovenous fistula. Neuroradiology 2001;43:784–786. Vera Zingler,

Iatrogenic vertebral arteriovenous fistula treated with a hemobahn stent-graft.

JV: Direct cervical vertebrovenous fistula with radiculopathy and MRI changes resolving after successful endovascular embolisation: A report of two cases.

Radiation-induced brachial plexopathy: Clinical and electromyographical (EMG) considerations in 13 cases. Electromyogr Clin Neurophysiol

http://epub.ub.uni-muenchen.de/16801/1/10_1159_000082370.pdf

Stent grafting resolved brachial plexus neuropathy due to cervical arteriovenous fistula

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