Brandt, T.

Brückmann, H.

Brüning, R.

Hamann, Gerhard F.

Strupp, M.

English

M. Strupp

H. Brückmann

G.F. Hamann

R. Brüning

T. Brandt

Crossref

Simultaneous Brachial Diplegia and Rotational Vertigo due to Combined Spinal Anterior and Vertebrobasilar Embolism

Universität München: Elektronischen Publikationen

240 Short Reports
Eur Neurol 2000;43:240–242
Simultaneous Brachial Diplegia and Rotational
Vertigo due to Combined Spinal Anterior and
Vertebrobasilar Embolism
M. Strupp a, H. Brückmann b, G.F. Hamann a, R. Brüningc,
T. Brandt a
Departments of a Neurology, b Neuroradiology, and
cRadiology, University of Munich, Klinikum Grosshadern,
Munich, Germany
Simultaneous ischemia of the vertebrobasilar and anterior spinal
artery territory is rare. Cheshire et al. [1] did not find evidence of
brainstem symptoms in any of their 44 patients with spinal cord
infarction nor in the additionally reviewed 155 cases. However, in
1966 Boudin et al. [2] reported on 2 patients with combined brain-
stem and spinal cord lesions who had vertebral-artery stenosis docu-
mented by angiography, and recently Pullicino [3] described a
patient with vertigo and bilateral distal upper limb amyotrophy. We
report on an exceptional case of combined, simultaneous ischemia in
the vertebrobasilar and anterior spinal artery territory.
Case Report
A 56-year-old woman without any risk factors for atherosclerosis
developed an acute case of severe rotatory vertigo, double vision and
initially complete paralysis of both arms accompanied by pain and
hypesthesia in both arms. The arm weakness considerably improved
in 1 day, while vertigo slowly disappeared over the following 10 days.
The patient was admitted to our hospital 8 days after symptom onset.
Clinical examination revealed (1) discrete central ocular and vestibu-
lar dysfunction with central downbeat positional nystagmus in head-
hanging position, saccadic vertical smooth pursuit and an upward
gaze-holding deficit; (2) a proximally accentuated moderate paresis
of both arms right/left: e.g. deltoid 4+/4–, triceps brachii 5/4+, and
biceps brachii muscle 5–/4; and (3) discrete paresthesia of the derma-
tomes of C5–C7 with intermittent proximally accentuated pain in the
left arm. The biceps jerk was absent on the left and attenuated on the
right. Extracranial Duplex sonography and MR angiography of the
vertebral and basilar arteries were normal. Transesophageal echocar-
diography showed a patent foramen ovale (PFO) with an atrial sep-
tum aneurysm (ASA) with spontaneous right-left shunting. Electro-
myography performed 6 weeks after symptom onset showed evi-
dence of denervation in the left deltoid and biceps muscles. The
patient has been receiving warfarin for 6 months now and has devel-
oped no further symptoms.
Cranial and Spinal MR Imaging. The initial cranial MRI per-
formed on admission (8 days after symptom onset) using T2-
weighted images showed a hyperintense lesion in the left cerebellar
hemisphere with focal contrast enhancement (fig. 1). Transversal spi-
nal T1-weighted sections at the level of C5 also showed contrast
enhancement with an ‘owl’s or snake-eyes’ pattern’, i.e. focal signal
abnormalities involving primarily the anterior horns of the gray mat-
ter (fig. 2). The transversal T2-weighted spinal images showed a
small, long hyperintense lesion in the anterior part of the cervical
cord from C3 to C6.
Discussion
The diagnosis of this very rare condition was clinically supported
by its acute onset combined with rotatory vertigo and simultaneous
Short Reports 241
Fig. 1. a Cranial axial MR images showed a
hyperintense lesion in the T2-weighted im-
ages (TR/TE = 4,800/78 ms, 5.0 mm slice
thickness, turbo spin echo, 512 ! 224 ma-
trix, 1 echo) in the left cerebellar hemisphere
(territory of the posterior inferior cerebellar
artery). b Cranial sagittal MRI. After ap-
plication of gadopentate dimeglumine (0.1
mmol/kg body weight) contrast enhance-
ment was observed in the T1-weighted im-
ages (TR/TE = 600/15 ms, 5.0 mm slice
thickness, 256 ! 224 matrix, 1 echo).
Fig. 2. a Initial transversal spinal T2-weighted images (TR/TE = 440/
17 ms, 3.0 mm slice thickness, 256 ! 224 matrix) demonstrated
bilateral symmetrical contrast enhancement in the anterio-lateral
parts of the spinal cord at the level of C5 after the application of gado-
pentate dimeglumine (owl’s or snake-eyes pattern). b Initial sagittal
spinal T1-weighted images (same sequence, 4.0 mm slice thickness)
also showed contrast enhancement in the anterior part of the cervical
cord at the level of C5 and C6.
flaccid brachial diplegia. The similarity of cranial and cervical T2-
weighted MR images as well as the contrast enhancement in the cere-
bellum and the cervical cord can be explained only by simultaneous
infarction. Of the 3 published cases only 1 has been documented by
MRI [3], and the combined lesion was due to vertebral dissection.
The pathomechanism also differed: in our patient, the most likely
cause was embolism, since the patient had a PFO, an ASA, no risk
factors for atherosclerosis and no evidence of vasculitis. Transversal
cervical MRI showed almost symmetrical, bilateral focal hyperinten-
sities in the anterior cord (owl’s or snake-eyes pattern) [4], which
occupied the highly vascularized anterior horn areas, near the border
zone between the anterior spinal artery and the pial artery plexus
territories (documented by Turnbull et al. [5]) and perimedullary net-
works (reported by Lazorthes [6]). The infarct was within the anterior
horn areas, which are supplied by terminal perforating arteries, espe-
cially the central artery with its ramifications within the gray matter;
a similar localization has been described by Pullicino [3]. The periph-
eral medullary area, however, is protected against ischemia by a sys-
tem of longitudinal anastomoses built by anterior and posterior spi-
nal arteries, and reinforced by various segmental levels. Further-
more, the MR images demonstrate that the anterior horn with the
motoneurons from C3 to C6 was primarily involved. This would also
correspond to the high metabolic demand of the motoneurons. Their
involvement explains the peripheral type of paraparesis of the upper
limbs, which mainly affected the left deltoid and biceps brachii mus-
cles and is compatible with the electromyographic findings of dener-
vation [7] on the left. In conclusion, infarction may have been limited
to the gray matter of the anterior horn due to its specific pattern of
arterial supply and great (metabolic) vulnerability to ischemia [8].
Acknowledgement
We thank Ms. J. Benson for copyediting the manuscript.
a b
a
b
242 Short Reports
References
1 Cheshire WP, Santos CC, Massey W, Howard JF: Spinal cord infarction:
Etiology and outcome. Neurology 1996;47:321–330.
2 Boudin G, Pepin B, Cassan JL, Vernant JC, Gazengel J: Medullary reper-
cussion of thromboses or stenoses of the vertebral artery (apropos of 3
cases). Rev Neurol (Paris) 1966;114:263–270.
3 Pullicino P: Bilateral distal upper limb amyotrophy and watershed infarcts
from vertebral dissection. Stroke 1994;25:1870–1872.
4 Al Mefty O, Harkey LH, Middleton TH, Smith RR, Fox JL: Myelopathic
cervical spondylotic lesions demonstrated by magnetic resonance imaging.
J Neurosurg 1988;68:217–222.
5 Turnbull IM, Brieg A, Hassler O: Blood supply of cervical spinal cord in
man. A microangiographic cadaver study. J Neurosurg 1966;24:951–965.
6 Lazorthes G: Pathology, classification and clinical aspects of vascular dis-
ease of the spinal cord; in Vinken PJ, Bruyn GW (eds): Handbook of Clini-
cal Neurology. Amsterdam, North-Holland, 1972, pp 492–506.
7 Levin KH, Daube JR: Spinal cord infarction: Another cause of ‘lumbosa-
cral polyradiculopathy’. Neurology 1984;34:389–390.
8 Gilles FH, Nag D: Vulnerability of human spinal cord in transient cardiac
arrest. Neurology 1971;21:833–839.
Priv.-Doz. Dr. Michael Strupp, MD, Department of Neurology
University of Munich, Klinikum Grosshadern
Marchioninistrasse 15, D–81366 Munich (Germany)
Tel. +49 89 7095 2571, Fax +49 89 7095 5584
E-Mail mstrupp@nefo.med.uni-muenchen.de


Simultaneous brachial diplegia and rotational vertigo due to combined spinal anterior and vertebrobasilar embolism

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

240 Short ReportsEur Neurol 2000;43:240–242Simultaneous Brachial Diplegia and RotationalVertigo due to Combined Spinal Anterior andVertebrobasilar EmbolismM. Strupp a, H. Brückmann b, G.F. Hamann a, R. Brüningc,T. Brandt aDepartments of a Neurology, b Neuroradiology, andcRadiology, University of Munich, Klinikum Grosshadern,Munich, GermanySimultaneous ischemia of the vertebrobasilar and anterior spinalartery territory is rare. Cheshire et al. [1] did not find evidence ofbrainstem symptoms in any of their 44 patients with spinal cordinfarction nor in the additionally reviewed 155 cases. However, in1966 Boudin et al. [2] reported on 2 patients with combined brain-stem and spinal cord lesions who had vertebral-artery stenosis docu-mented by angiography, and recently Pullicino [3] described apatient with vertigo and bilateral distal upper limb amyotrophy. Wereport on an exceptional case of combined, simultaneous ischemia inthe vertebrobasilar and anterior spinal artery territory.Case ReportA 56-year-old woman without any risk factors for atherosclerosisdeveloped an acute case of severe rotatory vertigo, double vision andinitially complete paralysis of both arms accompanied by pain andhypesthesia in both arms. The arm weakness considerably improvedin 1 day, while vertigo slowly disappeared over the following 10 days.The patient was admitted to our hospital 8 days after symptom onset.Clinical examination revealed (1) discrete central ocular and vestibu-lar dysfunction with central downbeat positional nystagmus in head-hanging position, saccadic vertical smooth pursuit and an upwardgaze-holding deficit; (2) a proximally accentuated moderate paresisof both arms right/left: e.g. deltoid 4+/4–, triceps brachii 5/4+, andbiceps brachii muscle 5–/4; and (3) discrete paresthesia of the derma-tomes of C5–C7 with intermittent proximally accentuated pain in theleft arm. The biceps jerk was absent on the left and attenuated on theright. Extracranial Duplex sonography and MR angiography of thevertebral and basilar arteries were normal. Transesophageal echocar-diography showed a patent foramen ovale (PFO) with an atrial sep-tum aneurysm (ASA) with spontaneous right-left shunting. Electro-myography performed 6 weeks after symptom onset showed evi-dence of denervation in the left deltoid and biceps muscles. Thepatient has been receiving warfarin for 6 months now and has devel-oped no further symptoms.Cranial and Spinal MR Imaging. The initial cranial MRI per-formed on admission (8 days after symptom onset) using T2-weighted images showed a hyperintense lesion in the left cerebellarhemisphere with focal contrast enhancement (fig. 1). Transversal spi-nal T1-weighted sections at the level of C5 also showed contrastenhancement with an ‘owl’s or snake-eyes’ pattern’, i.e. focal signalabnormalities involving primarily the anterior horns of the gray mat-ter (fig. 2). The transversal T2-weighted spinal images showed asmall, long hyperintense lesion in the anterior part of the cervicalcord from C3 to C6.DiscussionThe diagnosis of this very rare condition was clinically supportedby its acute onset combined with rotatory vertigo and simultaneousShort Reports 241Fig. 1. a Cranial axial MR images showed ahyperintense lesion in the T2-weighted im-ages (TR/TE = 4,800/78 ms, 5.0 mm slicethickness, turbo spin echo, 512 ! 224 ma-trix, 1 echo) in the left cerebellar hemisphere(territory of the posterior inferior cerebellarartery). b Cranial sagittal MRI. After ap-plication of gadopentate dimeglumine (0.1mmol/kg body weight) contrast enhance-ment was observed in the T1-weighted im-ages (TR/TE = 600/15 ms, 5.0 mm slicethickness, 256 ! 224 matrix, 1 echo).Fig. 2. a Initial transversal spinal T2-weighted images (TR/TE = 440/17 ms, 3.0 mm slice thickness, 256 ! 224 matrix) demonstratedbilateral symmetrical contrast enhancement in the anterio-lateralparts of the spinal cord at the level of C5 after the application of gado-pentate dimeglumine (owl’s or snake-eyes pattern). b Initial sagittalspinal T1-weighted images (same sequence, 4.0 mm slice thickness)also showed contrast enhancement in the anterior part of the cervicalcord at the level of C5 and C6.flaccid brachial diplegia. The similarity of cranial and cervical T2-weighted MR images as well as the contrast enhancement in the cere-bellum and the cervical cord can be explained only by simultaneousinfarction. Of the 3 published cases only 1 has been documented byMRI [3], and the combined lesion was due to vertebral dissection.The pathomechanism also differed: in our patient, the most likelycause was embolism, since the patient had a PFO, an ASA, no riskfactors for atherosclerosis and no evidence of vasculitis. Transversalcervical MRI showed almost symmetrical, bilateral focal hyperinten-sities in the anterior cord (owl’s or snake-eyes pattern) [4], whichoccupied the highly vascularized anterior horn areas, near the borderzone between the anterior spinal artery and the pial artery plexusterritories (documented by Turnbull et al. [5]) and perimedullary net-works (reported by Lazorthes [6]). The infarct was within the anteriorhorn areas, which are supplied by terminal perforating arteries, espe-cially the central artery with its ramifications within the gray matter;a similar localization has been described by Pullicino [3]. The periph-eral medullary area, however, is protected against ischemia by a sys-tem of longitudinal anastomoses built by anterior and posterior spi-nal arteries, and reinforced by various segmental levels. Further-more, the MR images demonstrate that the anterior horn with themotoneurons from C3 to C6 was primarily involved. This would alsocorrespond to the high metabolic demand of the motoneurons. Theirinvolvement explains the peripheral type of paraparesis of the upperlimbs, which mainly affected the left deltoid and biceps brachii mus-cles and is compatible with the electromyographic findings of dener-vation [7] on the left. In conclusion, infarction may have been limitedto the gray matter of the anterior horn due to its specific pattern ofarterial supply and great (metabolic) vulnerability to ischemia [8].AcknowledgementWe thank Ms. J. Benson for copyediting the manuscript.a bab242 Short ReportsReferences1 Cheshire WP, Santos CC, Massey W, Howard JF: Spinal cord infarction:Etiology and outcome. Neurology 1996;47:321–330.2 Boudin G, Pepin B, Cassan JL, Vernant JC, Gazengel J: Medullary reper-cussion of thromboses or stenoses of the vertebral artery (apropos of 3cases). Rev Neurol (Paris) 1966;114:263–270.3 Pullicino P: Bilateral distal upper limb amyotrophy and watershed infarctsfrom vertebral dissection. Stroke 1994;25:1870–1872.4 Al Mefty O, Harkey LH, Middleton TH, Smith RR, Fox JL: Myelopathiccervical spondylotic lesions demonstrated by magnetic resonance imaging.J Neurosurg 1988;68:217–222.5 Turnbull IM, Brieg A, Hassler O: Blood supply of cervical spinal cord inman. A microangiographic cadaver study. J Neurosurg 1966;24:951–965.6 Lazorthes G: Pathology, classification and clinical aspects of vascular dis-ease of the spinal cord; in Vinken PJ, Bruyn GW (eds): Handbook of Clini-cal Neurology. Amsterdam, North-Holland, 1972, pp 492–506.7 Levin KH, Daube JR: Spinal cord infarction: Another cause of ‘lumbosa-cral polyradiculopathy’. Neurology 1984;34:389–390.8 Gilles FH, Nag D: Vulnerability of human spinal cord in transient cardiacarrest. Neurology 1971;21:833–839.Priv.-Doz. Dr. Michael Strupp, MD, Department of NeurologyUniversity of Munich, Klinikum GrosshadernMarchioninistrasse 15, D–81366 Munich (Germany)Tel. +49 89 7095 2571, Fax +49 89 7095 5584E-Mail mstrupp@nefo.med.uni-muenchen.de

Bilateral distal upper limb amyotrophy and watershed infarcts from vertebral dissection. Stroke

classification and clinical aspects of vascular disease of the spinal cord;

Daube JR: Spinal cord infarction: Another cause of ‘lumbosacral polyradiculopathy’.

Fox JL: Myelopathic cervical spondylotic lesions demonstrated by magnetic resonance imaging.

JF: Spinal cord infarction: Etiology and outcome.

Medullary repercussion of thromboses or stenoses of the vertebral artery (apropos of 3 cases). Rev Neurol

O: Blood supply of cervical spinal cord in man. A microangiographic cadaver study.

Vulnerability of human spinal cord in transient cardiac arrest. Neurology 1971;21:833–839.

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Simultaneous brachial diplegia and rotational vertigo due to combined spinal anterior and vertebrobasilar embolism

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