Extracranial vertebral artery pseudoaneurysm presenting as subarachnoid hemorrhage.
S S Kaplan, C S Ogilvy, R Gonzalez, D Gress and J Pile-Spellman
Stroke. 1993;24:1397-1399
doi: 10.1161/01.STR.24.9.1397
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1397
Case Reports
Extracranial Vertebral Artery Pseudoaneurysm
Presenting as Subarachnoid Hemorrhage
Stuart S. Kaplan, BA; Christopher S. Ogilvy, MD; Ramon Gonzalez, MD;
Daryl Gress, MD; John Pile-Spellman, MD
Background and Purpose: True aneurysms of the extracranial vertebral artery are rare. The usual
pathogenesis of aneurysms in this location is either penetrating or blunt trauma with resultant
pseudoaneurysm formation. We report a postpartum patient with a presumed traumatic pseudoaneurysm
of the extracranial vertebral artery presenting with subarachnoid hemorrhage.
Case Description: A 41-year-old woman had three episodes of neck stiffness 1 month after an
uncomplicated vaginal delivery. The last episode, 3 days before admission, was accompanied by intense
neck and head pain and paresthesias that extended into the left arm, thumb, and forefinger.
Results: Lumbar puncture showed subarachnoid hemorrhage. Angiography revealed a left vertebral
artery dissection from C6-7 to C3 with pseudoaneurysm at C5-6. Computed tomography demonstrated
impingement of the C6 root at the foramen by this lesion. The lesion was successfully treated by balloon
occlusion of the vertebral artery.
Conclusions: We present a patient with an extracranial vertebral pseudoaneurysm with subarachnoid
hemorrhage and cervical root impingement. To our knowledge, this is the first case of such a lesion
presenting as subarachnoid hemorrhage. The lesion was successfully treated using endovascular
techniques. (Stroke. 1993;24:1397-1399.)
KEY WoRDs * embolization, therapeutic * subarachnoid hemorrhage * vertebral artery
Aneurysms of the extracranial vertebral arteries are
rare, with blunt or penetrating trauma the
usual cause.1-16 Their unusual occurrence is
thought to be due in part to the relatively protected
location of the vertebral artery coursing within the
foramina transversaria of the cervical vertebrae. When
lesions occur in the first and second portion of the
vertebral artery, they are usually caused by penetrating
trauma. In contrast, when pseudoaneurysms occur in
the third portion of the vertebral artery, they are usually
associated with blunt trauma or flexion-extension injuries of the neck. We describe a postpartum patient with
a presumed traumatic pseudoaneurysm of the extracranial vertebral artery presenting with subarachnoid hemorrhage. Angiography revealed a dissection of the vertebral artery from the C6-7 level to the C3 level with a
pseudoaneurysm located at C5-6 with impingement of
the C6 nerve root at the neural foramen. This lesion was
successfully treated with intravascular techniques of
balloon occlusion of the vessel without complications.
To our knowledge, the occurrence of subarachnoid
hemorrhage in association with an extracranial vertebral pseudoaneurysm has not been previously reported.
Details about the treatment of this lesion are discussed.
Materials and Methods
Case Report
A 41-year-old gravida 4, para 4 woman had three
episodes of neck stiffness 1 month after an uncomplicated vaginal delivery. Each episode occurred suddenly.
The first episode lasted 2 to 3 days and resolved
spontaneously. It was not associated with headache.
The second attack occurred approximately 5 days later
and again resolved spontaneously during several days.
This episode was associated with bilateral shoulder pain
with radiation of pain down the left arm with low back
pain. Four days before admission the patient had her
third episode that was accompanied by intense headache, fever to 101°F and worsening neck stiffness. She
also had paresthesias that extended into the left arm,
thumb, and forefinger. Head computed tomography at
another hospital showed no subarachnoid hemorrhage.
Lumbar puncture showed 120,000 red blood cells/mm3
(in the fourth tube), 2000 white blood cells/mm3 (76%
polys, 2% lymphs), glucose 15 mg/dL, and protein 128
mg/dL; serum glucose was 103 mg/dL. Neck and cerebral angiography demonstrated an abnormality at the
C5-6 level of the left vertebral artery. The patient was
transferred to our hospital for further evaluation and
treatment.
Received March 3, 1993; revision received April 27, 1993;
accepted April 27, 1993.
From the Neurosurgical Service (S.S.K., C.S.O.), Interventional
Neuroradiology (R.G., J.P.S.) and Clinical Neurology (D.G.),
Massachusetts General Hospital, Boston.
Correspondence to Christopher S. Ogilvy, MD, Neurosurgical
Service, Massachusetts General Hospital, Fruit Street, Boston,
MA 02114.
Results
On initial examination, the patient was afebrile with a
blood pressure of 160/84 and a heart rate of 84 beats per
minute. Neurological examination was notable solely for
nuchal rigidity. The rest of the neurological and general
physical examination was normal. Cervical computed
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1398
Stroke Vol 24, No 9 September 1993
neurological deficit. A control angiogram showed normal filling of the contralateral vertebral artery and
anterior spinal artery. The patient was neurologically
unchanged. She was maintained on intravenous heparin
with partial thromboplastin times maintained at twice
the normal level. Subsequently, systemic anticoagulation was maintained with oral warfarin. She was discharged from the hospital 1 week after the embolization
procedure.
FIG 1. Cervical computed tomography at the CS-6 level
demonstrates an abnormal left vertebral artery with the
appearance of a filling defect with enhancement on the left
(arrow) that encroaches on the left C5-6 neural foramen.
tomography showed an abnormal left vertebral artery
with the appearance of a filling defect with enhancement centered at the C5-6 level on the left that encroached on the left C5-6 neural foramen (Fig 1). A
head computed tomography showed no parenchymal
abnormalities and no subarachnoid blood. Repeat angiogram performed the next day revealed a left vertebral artery dissection from the region of C6-7 to C3 with
pseudoaneurysm at C5 (Fig 2).
We proceeded with balloon occlusion of the left
vertebral artery to reduce the threat of hemorrhage
from the pseudoaneurysm. The vertebral artery at C3 -4
was obliterated using two detachable latex balloons. At
C6-7 two additional detachable balloons were placed.
The patient tolerated the vertebral occlusion without
J1~~~~~~~~~~~~~~~~~~7
A.\
B
V}m
FIG 2. Left vertebral angiogram demonstrates a dissection
from the region of C6-7 to C3 with pseudoaneurysm at C5
(arrowhead). A, Lateral projection. B, Oblique projection. C,
AP projection.
Discussion
Aneurysms of the extracranial vertebral arteries are
rare because of their deeply protected location. They
are usually the result of penetrating or blunt trauma to
the neck.' 16 The trauma needed to produce a vertebral
pseudoaneurysm can be slight. Pseudoaneurysm formation has been reported following cervical dislocation,'7
cervical fracture,18 chiropractic manipulation.9'20 motor
vehicle accident,2' radon seed implantation,22 spinal
surgery,23 subelavian vein catheterization,24 cervicothoracic sympathectomy,25 cardiac pacemaker placement,26
and head trauma.20 Nontraumatic aneurysms as a whole
have been far less frequent. With regard to our patient,
abnormal movements in the neck during her prior
hospitalization and the forces associated with giving
birth may have caused the artery to stretch unduly with
resultant pseudoaneurysm formation and subarachnoid
hemorrhage.
It is difficult to diagnose a vertebral aneurysm from
clinical symptoms alone because the symptoms vary
according to the location of the lesion. A patient can
develop symptoms related to compression of surrounding structures or vertebrobasilar ischemic symptoms due
to thrombosis and embolization. The signs and symptoms can include pain in the neck or occipital region,
hearing difficulty, tinnitus, vertigo, nausea and vomiting, dysphagia, Horner's syndrome, sensory disturbances of the upper or lower extremities, and gait
disturbance. A large pseudoaneurysm with intraspinal
or intracranial extension can lead to cervical spinal cord
compression causing myelopathy27 or root compression
producing a radiculopathy.16 In addition, brain-stem
compression or lower cranial nerve compression can
occur.27 To our knowledge, this is the first patient with
an extracranial vertebral artery pseudoaneurysm presenting with subarachnoid hemorrhage confirmed by
lumbar puncture. Her cerebrospinal fluid showed a low
glucose for which we have no obvious explanation (all
cultures showed no growth of organisms).
Direct surgical approach to vertebral pseudoaneurysms has resulted in a high incidence of mortality and
morbidity. This is due to the artery's anatomic location,
extensive periarterial venous plexus, numerous sources
of collateral blood flow, and the risk of dislodging
emboli during manipulation.'3 27-30 The surgical technique of repairing an injury to the first three parts of the
vertebral artery has been outlined.15,31 Direct arterial
ligation, aneurysm trapping, balloon embolization, and
surgical revascularization have all been used and are
reviewed elsewhere.23 Other techniques that have been
described include direct vascular repair,23 aneurysmorrhaphy and direct closure,'4 and surgical repair using
catheters with balloons on the tips placed intraoperatively to occlude flow during the repair.26 In some
instances, the aneurysms have been left alone.17,19,20
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Kaplan et al Vertebral Pseudoaneurysm with SAH
Most vertebral artery pseudoaneurysms can be safely
treated with percutaneous transcatheter embolization.
Vertebral artery occlusion has been used with circulation to the basilar system maintained by the contralateral vertebral artery. The posterior inferior cerebellar
artery (PICA) on the side of the injury must fill via the
contralateral vertebral to avoid the possible development of a lateral medullary infarction. Test occlusion in
an awake patient is an advantage of endovascular
procedures. If occlusion is not tolerated, a bypass
procedure combined with vertebral artery ligation may
be considered.13 If occlusion is tolerated, systemic anticoagulation should be considered to avoid possible
thromboembolic complications. Although balloons leak
and collapse over time, they maintain their original
volume long enough to allow for formation of wellorganized thrombus.27,32
Primary repair of a vertebral pseudoaneurysm should
be reserved for unstable patients with active hemorrhage, large pseudoaneurysms that cannot be embolized, and in those instances when the contralateral
vertebral artery is either hypoplastic, absent, or terminates in the PICA.29,31 Verbral artery ligation is well
tolerated in most patients provided adequate collateral
exists through the contralateral vertebral artery.28 However, the presence of a patent contralateral vertebral
artery does not guarantee an adequate blood supply to
the basilar system during surgical manipulation as the
head may be sharply rotated and extended. This head
position can lead to occlusion of the contralateral
vertebral artery.33-35 For pseudoaneurysms of the vertebral artery, treatment should be considered early to
avoid lesion enlargement, hemorrhage, or distal embolization of thrombus.
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