Case Report

Recurrent Acute Ischemic Stroke Associated with Dolichoectatic Aneurysm of the Internal
Carotid Artery
Yusuke Morinaga, Kouhei Nii, Kimiya Sakamoto, Ritsurou Inoue, Takafumi Mitsutake, Hayatsura Hanada

- BACKGROUND: Cranial

Key words
- Acute ischemic stroke
- Best medical treatment
- DOAC
- Dolichoectatic aneurysm
- ICA
- Vertebrobasilar dolichoectasia

Abbreviations and Acronyms
ICA: Internal carotid artery
Department of Neurosurgery, Fukuoka University Chikushi
Hospital, Chikushino-shi, Japan
To whom correspondence should be addressed:
Yusuke Morinaga, M.D.
[E-mail: yu_the_morio@yahoo.co.jp]
Citation: World Neurosurg. (2019) 126:146-150.
https://doi.org/10.1016/j.wneu.2019.02.153
Journal homepage: www.journals.elsevier.com/worldneurosurgery
Available online: www.sciencedirect.com
1878-8750/$ - see front matter ª 2019 Elsevier Inc. All
rights reserved.

INTRODUCTION
Cranial
artery
dolichoectasia
is
characterized by dilated, elongated, and
tortuous cranial arteries such as the distal
internal
carotid
arteries
(ICAs).1
Dolichoectasia
and
dolichoectatic
aneurysm—particularly of the ICA—are
rare (prevalence 0.05%e0.06%)2,3 but
can lead to stroke. We report a case of
acute cerebral infarction associated with
a dolichoectatic aneurysm of the left ICA
combined with right dolichoectatic
aneurysm, vertebrobasilar dolichoectasia,
and atrial fibrillation. Written informed
consent was obtained from the patient
for publication of this case report and
the accompanying images, and the study
design was approved by the appropriate
ethics review board.

artery dolichoectasia is characterized by dilated,
elongated, and tortuous cranial arteries. Dolichoectasia and dolichoectatic
aneurysm—particularly of the internal carotid arteries (ICAs)—are rare but can
lead to stroke. We report a case of suspected recurrent acute cerebral infarction
and dolichoectatic aneurysm of the left ICA combined with right dolichoectatic
aneurysm, vertebrobasilar dolichoectasia, and atrial fibrillation.

- CASE DESCRIPTION: An 80-year-old man experienced 3 days of recurrent

aphasia episodes. He had a history of atrial fibrillation and hypertension.
Neurologic findings indicated Wernicke aphasia. Magnetic resonance imaging
showed acute cerebral infarction in the left temporal and parietal lobes. Magnetic resonance angiography and cerebral angiography showed dolichoectatic
aneurysms in the C1-2 portion of the bilateral ICAs and vertebrobasilar dolichoectasia. Possible diagnoses included convulsion, cardioembolic infarction,
atherothrombotic cerebral infarction, and acute ischemic stroke associated with
the dolichoectatic aneurysm of the left ICA. Oral antiplatelet drugs, direct oral
anticoagulants, and anticonvulsants were administered. Nine months after
initial hospitalization, the patient had a recurrence of acute ischemic stroke and
was rehospitalized. He was treated with a new direct oral anticoagulant, clopidogrel, statins, and ethyl icosapentate. At 2 years after initial hospitalization,
he has had no recurrence of stroke, has a modified Rankin Scale score of 2, and
is currently being followed as an outpatient.
- CONCLUSIONS: A direct oral anticoagulant (edoxaban) and clopidogrel were

effective for suspected recurrent acute ischemic stroke associated with dolichoectatic aneurysm of the ICA combined with contralateral dolichoectatic
aneurysm, vertebrobasilar dolichoectasia, and atrial fibrillation.

CASE DESCRIPTION

antihypertensive medication and had good
compliance.
Neurologic
assessment
revealed Wernicke aphasia but no
apparent quadriplegia or other neurologic
deficits. The patient scored 6/42 on the
National Institutes of Health Stroke Scale.
Blood analysis (including blood cell
counts, biochemistry, and coagulation
parameters) revealed no abnormalities.
Electrocardiography revealed no atrial
fibrillation at hospitalization. Cardiac
ultrasound examination showed no intraatrial thrombus.

An 80-year-old man had episodic aphasia
for a duration of 3 days. He had a history
of hypertension and atrial fibrillation. He
was taking rivaroxaban (10 mg/day) and

Imaging Findings
Head computed tomography showed
marked elongation, dilation (>4.5 mm),

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and calcification from the left vertebral
artery to the basilar artery. Both ICAs were
dilated, elongated, and partially calcified;
the vascular lumens were uniform with
slightly increased density (Figure 1A).
Diffusion-weighted magnetic resonance
imaging showed areas of high intensity
from the left temporal lobe to the parietal
lobe consistent with the vessel-dominant
areas of the M2 inferior trunk
(Figure
1B).
Magnetic
resonance
angiography revealed aneurysms in the
C1-2 portion of both ICAs and poor visualization of both middle cerebral arteries
(Figure 1C). Time-of-flight magnetic
resonance angiography showed heterogeneous high-intensity changes from the left

WORLD NEUROSURGERY, https://doi.org/10.1016/j.wneu.2019.02.153

CASE REPORT
YUSUKE MORINAGA ET AL.

ACUTE ISCHEMIC STROKE AND DOLICHOECTATIC ANEURYSM

Figure 1. (A) Head computed tomography
images showing remarkable elongation,
dilation, and calcification from the left vertebral
artery to the basilar artery. The vascular lumen
was uniform with slightly increased density.
Both internal carotid arteries (ICAs) also
showed expansion with calcification. The
vascular lumens were uniform with slightly
increased density. (B) Diffusion-weighted
magnetic resonance imaging showing
high-intensity areas in the left temporal lobe

vertebral artery to the basilar artery and in
both ICAs (Figure 1D).
Acute cerebral infarction and thrombus
progression was assessed by cerebral
angiography. Giant cerebral ICA aneurysms at the C1-2 level (right, approximately 20 mm; left, approximately 25 mm;
with both anterior choroidal arteries
diverging from the aneurysms) were

and parietal lobe, consistent with the
vessel-dominant areas of the M2 inferior trunk
of the left middle cerebral artery. (C) Magnetic
resonance angiography showing aneurysms in
the C1-2 portion of the bilateral ICAs, with poor
visualization of the bilateral middle cerebral
arteries. (D) Time-of-flight magnetic resonance
angiography showing heterogeneous
high-intensity changes from the left vertebral
artery to the basilar artery and in the bilateral
ICAs.

found. Dolichoectatic aneurysms of the
bilateral ICAs were diagnosed (Figure 2A
and B). Aneurysmal blood flow was
turbulent and delayed bilaterally. Left
ICA angiography poorly visualized the
M2 inferior trunk from the origin, but
aneurysmal thrombosis was absent.
Right ICA angiography showed crossflow to the left ICA and left middle

WORLD NEUROSURGERY 126: 146-150, JUNE 2019

cerebral artery in the late arterial phase
during the Matas test and right M2 superior trunk stenosis. Bilateral cervical ICAs
were elongated but without severe stenosis. Bilateral external carotid artery angiography revealed an absence of
intracranial anastomosis. Vertebral angiography revealed significant elongation
and dilation from the left vertebral artery

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CASE REPORT
YUSUKE MORINAGA ET AL.

ACUTE ISCHEMIC STROKE AND DOLICHOECTATIC ANEURYSM

Figure 1. Continued.

to the basilar artery and dolichoectasia
(Figure 2C and D). Allcock test revealed no
cross-flow to the anterior circulatory
system.
Posthospitalization Course
The patient was treated with argatroban
by intravenous injection, an oral antiplatelet drug (aspirin, 100 mg/day), an
anticoagulant (rivaroxaban, 10 mg/day),
and an anticonvulsant (levetiracetam,
1000 mg/day). Electroencephalograms
conducted during oral administration of
the antiepileptic drugs did not show
epileptic waves. During hospitalization,
the patient did not have progressive cerebral infarction or a seizure attack. One
month after hospitalization, his aphasia
had subsided, and he was transferred to a
rehabilitation hospital. At this time, he
had a modified Rankin Scale score of 2.
Nine months after his initial hospitalization, the patient had mild right upper
limb paralysis and was transported to our
hospital again. His oral compliance was
good. Magnetic resonance imaging

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showed acute lacunar infarction in the left
centrum semiovale (Figure 3A and B). He
was
treated
with
edaravone
by
intravenous injection, an oral antiplatelet
drug (clopidogrel, 75 mg/day), an
anticoagulant (edoxaban tosylate hydrate,
30 mg/day), rosuvastatin calcium (2.5
mg/day), and ethyl icosapentate (1800
mg/day). The antithrombotic drugs
administered at the time of the patient’s
initial discharge were changed. The
patient’s right upper limb paralysis
improved, and he was discharged 1 week
after his second hospitalization. Two
years have passed since the patient’s
initial hospitalization. He has not been
readmitted for the recurrence of stroke;
however, he has aftereffects of aphasia
with a modified Rankin Scale score of 2,
and is currently being followed as an
outpatient.
DISCUSSION
The main causes of dolichoectasia include
atherosclerosis and hypertension,3 although

other conditions have been reported.4-6
From
a
pathogenic
perspective,
dolichoectasia is a dilatative arteriopathy
that occurs as a result of deficiencies in the
muscularis and internal elastic lamina,
Marfan
syndrome,
Ehlers-Danlos
syndrome, pseudoxanthoma elasticum,
and other less well-defined connective
tissue disorders.7 The patient in our case
had a history of chronic hypertension but
no family history of Marfan syndrome,
Ehlers-Danlos syndrome, or other relevant
pathologies. Therefore, atherosclerotic
vertebrobasilar
dolichoectasia
and
dolichoectatic aneurysms of both ICAs
were
possible
diagnoses.
Hyperimmunoglobulinemia
syndrome8
and infection9-11 may also cause arterial
dilation, but the patient’s IgE level was not
elevated, and there was no evidence of
infection. Dolichoectasia involving both
the vertebrobasilar and the carotid systems
is rare.1 Our case represents a particularly
rare manifestation, as dolichoectasia
existed in both the anterior and the
posterior circulation, with dolichoectatic
aneurysms of the bilateral intracranial
ICAs. Coupled with the absence of crossflow from the contralateral ICA and posterior circulation, the flow velocity of the left
middle cerebral artery was likely further
decreased, possibly contributing to cerebral
infarction.
The delayed flow velocity in our case
may be related to thrombus formation.
Transcranial Doppler studies of intracranial dolichoectasia have demonstrated
reduced peak flow velocity.12 Reduced flow
can lead to stagnation of the blood column
to give spontaneous echo contrast and
increase the risk of formation of a
thrombus with embolization distally.13
Our
patient
exhibited
increasing
symptoms similar to a transient ischemic
attack before hospitalization, suggesting
convulsion, a cardioembolic infarction, a
hemodynamic cerebral infarction, or an
artery-to-artery embolization mechanism,
all of which are associated with dolichoectatic aneurysms of the left ICA and/
or an atherothrombotic cerebral infarction
in the M2 inferior trunk.
Previously, in dolichoectasia cases,
anticoagulant therapy was recommended

WORLD NEUROSURGERY, https://doi.org/10.1016/j.wneu.2019.02.153

CASE REPORT
YUSUKE MORINAGA ET AL.

ACUTE ISCHEMIC STROKE AND DOLICHOECTATIC ANEURYSM

Figure 2. (A) Angiography of the left internal
carotid artery (ICA) showing giant cerebral
aneurysms at the C1-2 portion (25 mm on the
left side), with left anterior choroidal artery
diverging from the aneurysm. This finding
indicated a dolichoectatic aneurysm of the left
ICA. Left ICA angiography also showed poor
visualization of the M2 inferior trunk of the left
middle cerebral artery from the origin and no
thrombosis in the left ICA aneurysm. (B)
Angiography of the right ICA showing giant

for the prevention of thrombi and embolisms when ischemic symptoms
occurred14; antiplatelet therapy was
always recommended, regardless of
ischemia.15 In our case, given the
standard treatment for atherothrombotic
cerebral infarction and the patient’s
atrial fibrillation, antiplatelet therapy

cerebral aneurysms at the C1-2 portion (20
mm on the right side), with right anterior
choroidal artery diverging from the aneurysm.
This finding indicated a dolichoectatic
aneurysm of the right ICA. Right ICA
angiography also showed right M2 superior
trunk stenosis. (C and D) Left vertebral
angiography showing significant elongation
and dilation from the left vertebral artery to the
basilar artery with findings of dolichoectasia.

and
anticoagulant
therapy
were
administered
during
his
initial
hospitalization.
Although
these
therapies did not resolve his aphasia,
they may have prevented progression of
cerebral infarction during his initial
hospitalization. Although there is no
evidence for dolichoectasia causing

WORLD NEUROSURGERY 126: 146-150, JUNE 2019

ischemic strokes, we treated the patient
using the best medical treatment. The
patient was given a direct oral
anticoagulant (edoxaban), clopidogrel,
statins, and ethyl icosapentate following
the recurrence of ischemic stroke,
which was based on the mechanism of
thrombi formation.

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CASE REPORT
YUSUKE MORINAGA ET AL.

ACUTE ISCHEMIC STROKE AND DOLICHOECTATIC ANEURYSM

Figure 3. (A) Diffusion-weighted magnetic
resonance imaging showing a high-intensity
spot in the left centrum semiovale. (B)
Magnetic resonance angiography at the

CONCLUSIONS
We report an extremely rare case of suspected recurrent acute cerebral infarction
associated with a dolichoectatic aneurysm
of the ICA combined with dolichoectatic
aneurysm of the contralateral ICA, vertebrobasilar dolichoectasia, and atrial
fibrillation. Standard medical care, which
included a direct oral anticoagulant
(edoxaban), clopidogrel, statins, and ethyl
icosapentate, was effective for our patient.

second hospitalization showing aneurysms in
the C1-2 portions of the bilateral internal
carotid arteries, with poor visualization of the
bilateral middle cerebral arteries.

4. Schoolman A, Kepes JJ. Bilateral spontaneous
carotid-cavernous fistulae in Ehlers-Danlos syndrome: case report. J Neurosurg. 1967;26:82-86.
5. Finney LH, Roberts TS, Anderson RE. Giant
intracranial aneurysm associated with Marfan’s
syndrome. Case report. J Neurosurg. 1976;45:
342-347.
6. Blumenkopf B, Huggins MJ. Tuberous sclerosis
and multiple intracranial aneurysms: case report.
Neurosurgery. 1985;17:797-800.
7. Sadahiro H, Ishihara H, Goto H, et al. Repeated
cerebral ischemia caused by extracranial carotid
artery dolichoectasia. J Neuroimaging. 2014;24:
83-87.

12. Hennerici M, Rautenberg W, Schwartz A.
Transcranial Doppler ultrasound for the assessment of intracranial arterial flow velocity—Part 2.
Evaluation of intracranial arterial disease. Surg
Neurol. 1987;27:523-532.

13. De Georgia M, Belden J, Pao L, Pessin M, Kwan E,
Caplan L. Thrombus in vertebrobasilar
dolichoectatic artery treated with intravenous
urokinase. Cerebrovasc Dis. 1999;9:28-33.

14. Echiverri HC, Rubino FA, Gupta SR, Gujrati M.
Fusiform aneurysm of the vertebrobasilar arterial
system. Stroke. 1989;20:1741-1747.

ACKNOWLEDGMENTS
We thank Dr. Ayumu Eto and Dr. Fumihiro Hiraoka for helpful discussion
regarding our case.
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Conflict of interest statement: The authors declare that the
article content was composed on the absence of any
commercial or financial relationships that could be construed
as a potential conflict of interest.
Received 10 January 2019; accepted 22 February 2019
Citation: World Neurosurg. (2019) 126:146-150.
https://doi.org/10.1016/j.wneu.2019.02.153
Journal homepage: www.journals.elsevier.com/worldneurosurgery
Available online: www.sciencedirect.com
1878-8750/$ - see front matter ª 2019 Elsevier Inc. All
rights reserved.

WORLD NEUROSURGERY, https://doi.org/10.1016/j.wneu.2019.02.153