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Cervical Rib, a Rare Cause of Recurrent Stroke in the Young Case Report
Article in The Neurologist · September 2012
DOI: 10.1097/NRL.0b013e31826754a9 · Source: PubMed
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CASE REPORT/CASE SERIES
Cervical Rib, a Rare Cause of Recurrent Stroke in the Young
Case Report
Rashim Kataria, MS, MCh, Arun Sharma, MS, Trilochan Srivastava, MD, DM,
Hardev Bagaria, MS, MCh, and Anil Sharma, MS, MCh
Background: Cervical rib usually causes neurological symptoms in
the upper limb but stroke as an initial presentation is very uncommon.
Recurrent supratentorial and infratentorial stroke in a single patient is
very rare. Cervical rib can lead to anterograde or retrograde thromboembolic phenomenon leading to ischemic stroke.
Case Report: A 14-year-old girl presented with a history of sudden
onset of loss of consciousness and left hemiparesis of 2 days duration.
She had a similar episode 2 years ago, from which she had recovered to
a large extent with minimal residual left hemiparesis. On examination,
she was unconscious and localizing to pain on the right side. Radial
pulse was absent on the right side. Magnetic resonance imaging of the
brain revealed a right fronto-temporo-parietal old infarct with a new
subacute infarct involving right cerebellar hemisphere and brainstem.
X-ray of the chest showed a right-sided cervical rib. Computed tomographic angiography of the neck vessels revealed stenosis of subclavian
artery at the site of the cervical rib with poststenosis dilatation. Patient
was managed with anticoagulant and antiplatelet therapy initially and
excision of the cervical rib was performed as a definitive procedure. She
responded well to the treatment and at 6 months of follow-up, the
strength on the left side had improved substantially. She was capable of
doing her daily activities independently with little imbalance.
Conclusions: The reported patient is the first in the literature who
suffered recurrent supratentorial and infratentorial stroke as a complication of cervical rib. We stress the need for early diagnosis of this
easily treatable cause of stroke in the young.
Key Words: cervical rib, thoracic outlet syndrome (TOS), stroke,
thromboembolism
(The Neurologist 2012;18:321–323)
C
ervical rib is a supernumerary rib arising from the seventh
cervical vertebra. It is present in <1% of the normal
population and 50% to 80% are bilateral. It is usually
asymptomatic with only 10% to 20% of patients having
symptoms. The female to male ratio is 2:1.1 It can cause
thoracic outlet syndrome (TOS) as a result of entrapment of the
lower trunk of brachial plexus or the subclavian vessels. Stroke
can occur as a result of retrograde or anterograde, artery to
artery thromboembolic phenomenon.2–7 Chronic trauma to the
subclavian artery by the cervical rib leads to stenosis at the site
of the compression with poststenosis dilatation of the vessel,
From the Department of Neurosurgery, SMS Medical College, Jaipur,
Rajasthan, India.
The authors declare no conflict of interest.
Reprints: Rashim Kataria, MS, MCh, N-6 Gandhinagar MLA Flats, Behind
Jawahar Kala Kendra, Jaipur, 302015 Rajasthan, India. E-mail:
rashim_kat
@yahoo.com.
Copyright r 2012 by Lippincott Williams & Wilkins
ISSN: 1074-7931/12/1805-0321
DOI: 10.1097/NRL.0b013e31826754a9
The Neurologist
Volume 18, Number 5, September 2012
which in turn leads to the formation of a thrombus. During
overhead abduction of the arm, cervical rib completely
occludes the artery and there is retrograde flow from the subclavian artery to the common carotid and vertebral arteries
leading to retrograde thromboembolic phenomenon. In some
cases, the thrombus can extend proximally to involve the ostia
of the common carotid and vertebral arteries, thus leading to
antegrade thromboembolic phenomenon as well.4
CASE SUMMARY
A 14-year-old normotensive, nondiabetic girl presented with a
history of sudden onset of loss of consciousness and left hemiparesis of 2
days duration. She had a history of a similar episode 2 years ago, from
which she had recovered to large extent with minimal residual left
hemiparesis. She was not taking any medications for the last 1½ years and
had not followed up with her neurologist during that period. She did not
have any previous medical records or imaging with her when she presented to us. There was no history of recurrent right upper limb pain,
paresthesias, or weakness. On examination, she was unconscious and was
localizing to pain on the right side. Pupils were 3 mm in diameter and
reacted to light bilaterally. Blood pressure was 130/90 mm Hg, pulse rate
was 90 beats/min, and the radial pulse was absent on the right side.
Magnetic resonance imaging of the brain (Fig. 1) revealed an
area of old infarct involving the right fronto-temporo-parietal region
with dilatation of ipsilateral lateral ventricle due to volume loss. There
was a subacute infarct involving the right cerebellar hemisphere and
brainstem. Patient was thoroughly investigated for causes of recurrent
infarct in the young. An x-ray of the chest (Fig. 2) showed a right-sided
cervical rib. Computed tomographic angiography of the neck vessels
(Fig. 3) revealed a stenosis of the subclavian artery at the site of the
cervical rib, with poststenosis dilatation. A complete cutoff was seen at
the site of the cervical rib in overhead abduction of the right arm, with
refilling of the axillary artery by collaterals. She was initially managed
with anticoagulant and antiplatelet therapy and excision of the cervical
rib was performed as a definitive procedure later. She responded well
to the treatment and at 6-month follow-up, the strength on the left side
had improved substantially. She was capable of doing her daily
activities independently with little imbalance.
DISCUSSION
Symptomatic arterial compression from a cervical rib is
uncommon. Compression of the other 2 components in the
thoracic outlet, namely brachial plexus and subclavian vein, is
relatively more common. Arterial and venous TOS are usually
not difficult to recognize and the diagnosis can be confirmed
by angiography. The diagnosis of neurogenic TOS is somewhat more challenging because its symptoms of nerve compression are not unique.5 It has been observed that short and
incomplete ribs usually produce neurological complications by
nerve compression, whereas long or complete ribs as seen in
this patient present more often with arterial symptoms.
Thromboembolism of the forearm and digits is the most typical
clinical presentation of patients with distal subclavian artery
disease due to TOS. This was evident in the present case by the
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Kataria et al
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Volume 18, Number 5, September 2012
FIGURE 1. A, Axial T2-weighted MRI of the brain showing areas of old infarct involving the right MCA territory with dilatation of
ipsilateral lateral ventricle due to volume loss. B, Axial T2-weighted of the brain showing subacute infarct (arrow) involving right
cerebellar hemisphere. MCA indicates middle cerebral artery; MRI, magnetic resonance imaging.
absence of right radial pulse without significant ischemic
symptoms. Because of collateral formation, the ischemic
symptoms may be mild in many patients and this could be one
of the reasons of late presentation to the hospital. The presence
of collaterals also suggests longstanding stenosis and/or
thrombosis of the distal subclavian artery.8 Right carotid artery
stroke is more often described than vertebrobasilar stroke in
patients with a cervical rib. This phenomenon is possibly due
to the anatomic characteristics of the right carotid artery
branching from the brachiocephalic artery, and the differences
in caliber of the common carotid and vertebral arteries with
reduced resistance seen in the larger common carotid artery. It
is interesting to note that supratentorial strokes have always
been described with a right-sided cervical rib, whereas the
vertebrobasilar strokes have always been described with a
left-sided cervical rib. Left-sided carotid stroke has not been
described with cervical rib as left carotid arises from arch of
aorta.3 In the present case, the probable cause of the infratentorial infarct seems to be antegrade thromboembolic phenomenon from subclavian to right vertebral artery. Staging of
the stenosis of the subclavian artery and the surgical guidelines
based on these staging were given by Scher et.al.9 Stage I
FIGURE 2. PA view of chest x-ray showing a well formed rightsided cervical rib (arrow). PA indicates Posterio-anterior view.
322 | www.theneurologist.org
lesions have only arterial stenosis and minor poststenotic
dilatation and are managed by thoracic outlet decompression,
FIGURE 3. Computed tomographic angiogram of the neck vessels (with arm abducted) showing stenosis of right subclavian
artery (hollow black arrow) at the site of cervical rib with poststenotic dilatation (black arrow). Right vertebral is seen originating (white arrow) from the dilated segment.
r
2012 Lippincott Williams & Wilkins
The Neurologist
Volume 18, Number 5, September 2012
usually consisting of cervical rib resection. Stage II lesions
have intrinsic arterial damage usually with subclavian aneurysm formation and require rib resection, aneurysmectomy, and
arterial reconstruction. Stage III lesions present with distal
thromboembolic complications and require thrombectomy or
embolectomy in addition to thoracic outlet decompression and
arterial reconstruction.9 Transaxillary excision of the first rib is
a surgical procedure associated with very low morbidity rate
and excellent relief of symptoms. It can therefore be offered as
an early option for patients with TOS. This may be combined
with the supraclavicular approach if exposure of the subclavian
artery is required for vascular reconstruction. An early diagnosis and timely management of the cervical rib can prevent
life-threatening strokes in the young.
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Cervical Rib Presenting as Recurrent Stroke
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