ELSEVIER

SYSTEMIC MULTIPLE ANEURYSMS
OF
THE INTRACRANIAL
ARTERIES AND
VISCERAL ARTERIES:
CASE REPORT
Takahisa Fuse, M.D.,* ** Takuji Takagi,** Kazuo Yamada,* and Tsuneyuki Fukushima* *
*Department of Neurosurgery, Nagoya City University School of Medicine, Nagoya, Japan;
and **Depar?ment of Neurosurgery, Nagoya City Higashi General Hospital, Nagoya, Japan

Fuse T, Takagi T, Yamada K, Fukushima T. Systemic multiple
aneurysms of the intracranial arteries and viscera1arteries. Surg
Neurot 1996;46:258-62.
BACKGROUND

Systemic multiple aneurysms are rare, and an association
between intracranial aneurysms and visceral aneurysms
is an extremely rare occurrence.
CASE

PRESENTATION

This 56year-old woman was referred to our hospital with
subarachnoid hemorrhage. Physical examination revealed no particular morphologic features suggesting
connective tissue diseases. Cerebral angiography demonstrated two aneurysms of the left internal carotid artery
and the right middle cerebral artery. The internal carotid
artery aneurysm was occluded surgically. Four days later,
she had right hemiparesis and mild aphasia due to cerebral vasospasm. While she was started on hemodynamic
therapy and the administration of thromboxane A2 synthetase inhibitor for cerebral vasospasm, she suffered
intraabdominal hemorrhage due to the rupture of the
visceral artery aneurysms, and was treated with emergency surgery.
CONCLUSION

The hemodynamic therapy and the administration of
thromboxane A2 synthetase inhibitor for symptomatic
vasospasm may have precipitated the rupture of the intraabdominal aneurysm.
KEY

hibitor for cerebral vasospasm postoperatively,
she
suffered intraabdominal
hemorrhage
due to the
rupture of the visceral artery aneurysms. We describe the clinical findings and discuss the etiologic
consideration
in this extremely rare case, with a
review of the literature.

WORDS

Intracranial artery aneurysm, visceral artery aneurysm,
thromboxane A2 synthetase inhibitor.

S

ystemic multiple aneurysms are extremely
rare [7]. In this case, we describe a patient
with aneurysms of the internal carotid artery, the
middle cerebral artery, the gastroepiploic
artery,
and the bilateral gastric arteries. While undergoing
hypervolemic
and hypertensive
therapy and the
administration
of thromboxane
A2 synthetase inAddress reprint requests to: Takahisa Fuse, M.D., Department
of Neurosurgery,
Nagoya City University
School of Medicine,
1-Kawasumi,
Mizuho-cho,
Mizuho-ku,
Nagoya, 467 Japan.
Received October 2, 1995; accepted February
14, 1996.
0090-3019/96/$15.00
PIf SOO90-3019(96)00217-O

CASE

REPORT

This patient, a 56-year-old woman, suddenly developed a severe headache on the evening of March 8,
1995. She was admitted to our clinic at the Nagoya
City Higashi General Hospital, Japan, where she had
a stiff neck but was neurologically normal (Hunt and
Hess Grade 2). Blood pressure was elevated at
180-90 mm Hg. She had no past history of significant headaches or any other neurologic disturbances, and there was no family history of neurologic disease. Physical examination
revealed no
particular morphologic
features, suggesting such
connective tissue diseases as Ehlers-Danlos syndrome or Marfan’s syndrome. A CT scan revealed
evidence of a subarachnoid
hemorrhage
(SAH),
showing a predominantly
left-sided clot in the sylvian fissure (Figure 1). Cerebral angiograms were
obtained on March 9th. The left carotid angiogram
showed a saccular aneurysm at the Cl portion of
the internal carotid artery (lCA) and at the Ml-M2
bifurcation
of the right middle cerebral artery
(MCA) (Figure 2). The cause of the subarachnoid
hemorrhage in this patient was speculated to be the
rupture of the left ICA aneurysm.
On March 10, 1995, surgical treatment was carried out under general anesthesia. A left pterional
approach revealed an aneurysm of the carotid artery with a smooth, thin wall, which was clipped
with a straight clip. The patient’s recuperation was
uneventful until the fourth postoperative
day, when
655 Avenue

@ 1996 by Elsevier Science Inc.
of the Americas,
New York, NY 10010

Systemic Aneurysms of the Intracranial and Visceral Arteries

Surg Neurol
199ti46258-62

259

carotid angiography shows a saccular aneuqand aLeft
rysm at the C2 portion of left internal carotid artery
saccular aneurysm at the Ml-M2 bifurcation of the
right middle cerebral artery.
tomographic scan of the head demonIIsylvianComputed
strating subarachnoid hemorrhage mainly in the left
fissure.

she had right hemiparesis and mild aphasia, which
were considered to be due to cerebral vasospasm.
She was started on a dopamine hydrochloride
infusion and given 5% albumin to maintain a systolic
blood pressure of 170-190 mmHg, and was administered 80 mg of thromboxane
A2 synthetase inhibitor; sodium ozagrel. The right motor weakness and
aphasia resolved completely on the seventh postoperative day. On March 23, 1995 left carotid angiograms demonstrated
that the aneurysm of the left
ICA was completely occluded surgically, although
there was stenosis of the Cl portion of the carotid
artery associated with late vasospasm. Therefore,
intravascular
volume expansion, induced arterial
hypertension, and administration
of sodium ozagrel
were continued.
On March 24, 1995, the patient suddenly developed severe abdominal pain. Abdominal CT disclosed bleeding in the abdominal cavity (Figure 3)
and celiac arteriography
revealed several aneurysms in the gastroepiploic
and bilateral gastric
arteries (Pigure 4). An emergency exploratory laparotomy was carried out under general anesthesia,
demonstrating
a rupture of the gastroepiploic
artery aneurysms. The aneurysms were resected with
ligation of the gastroepiploic
and gastric arteries.
The postoperative
course was uneventful. On May
lst, the patient was discharged from the hospital
without neurologic deficits.

DISCUSSION
Cerebral vasospasm is a major cause of complications following subarachnoid hemorrhage. Progressive neurologic deterioration
occurs in patients after subarachnoid hemorrhage with a frequency that
ranges from 16%-66%, and is conventionally
presumed to be related to the luminal narrowing of the
major cerebral arteries due to cerebral vasospasm
and a decrease in cerebral blood flow below critical
levels, resulting in ischemia [4,5]. The accepted
therapeutic approach to such cerebral vasospasminduced deficits includes hemodilution,
hypertension, and hypervolemia.
Calcium-channel
blockers
and thromboxane
A2 synthetase inhibitor are also
an additional component of standard care, although
complications
experienced during therapy have included pulmonary edema, aneurysmal rebleeding,
myocardiac infarction, dilutional hyponatremia,
coagulopathy, hemothorax,
and intracranial
hemorrhage [5,11].
Our patient suffered an unusual complication
insofar as therapy for vasospasm is concerned: intraabdominal hemorrhage, resulting from the rupture
of visceral artery aneurysms. Aneurysms involving
the visceral arteries are uncommon, and about 50%
of those aneurysms occur in the splenic artery,
followed by 20% in the hepatic artery [ 1,101. These
aneurysms are insidious or are detected by chance,
and in many cases, their presence becomes evident
only at the time of rupture [2]. However, gastric
artery aneurysms or gastroepiploic
artery aneurysms are extremely rare and constitute only 4.9%
of the number of the visceral artery aneurysms [lo].

260

Surg Neural
1996;46:258-62

Fuse et al

Computed
tomographic
qonstrates
scan of the abdomen dembleeding in the abdominal cavity.

A review of the literature demonstrates
that an
association
between intracranial
aneurysms and
visceral aneurysms is an extremely rare occurrence, and the occurrence of multiple systemic aneurysms is almost always associated with collagen
tissue diseases, such as in Ehlers-Danlos syndrome
[9] or Marfan’s syndrome [ 131. In this case, until the
visceral artery aneurysms ruptured, we had not
realized that this patient had multiple systemic aneurysms. So the extraction of the tissue of the intracranial
aneurysm for pathologic
examination
was not performed, and this patient was not exam-

view of the celiac artery angiograqtery,‘ Anteroposterior
phy shows an aneurysm of the gastroepiploic arof the right gastric artery and of the left gastric
artery. These aneurysms are shaped like a dissecting
aneurysm.

ined in this regard. However, physical examination
in our case revealed no particular morphologic features suggesting such connective tissue diseases,
so we speculate that our patient had a chance occurrence of a coexisting abdominal aneurysm. However, a congenital cause for such multiple aneurysms cannot be excluded
because systemic
multiple aneurysms have been described in association with a variety of inherited connective-tissue
defects, and Niel-Dwyer et al reported that 11 of 17
patients associated with cerebral aneurysms were
Type III collagen-deficient
[ 81.
To the best of our knowledge, only one case has
been reported in the literature of intracranial aneurysms and visceral artery aneurysms with no apparent underlying disease. Kubo et al reported the
first case with aneurysms of the extracranial carotid artery, the intracranial vertebral artery, the
hepatic artery, and the splenic artery [6]. In their
case, the extracranial carotid artery aneurysm was
successfully resected, with end-to-end anastomosis
of the internal carotid artery. Five days later, an
aneurysm of the hepatic artery ruptured unexpectedly without hemodynamic
therapy (hypertension
and hypervolemia for cerebral vasospasm) or anticoagulative therapy,
Rupture of an abdominal vessel aneurysm after
an unrelated
surgical procedure
is uncommon.
Durham et al reported rupture of an abdominal
aortic aneurysm in one of 33 patients with a known
aortic aneurysm after. unrelated surgery [3]. They
speculated that an unrelated surgical intervention
hastens the time of rupture of an abdominal aortic
aneurysm, and that fear of postoperative
aneurysm

Systemic Aneurysms of the Intracranial and Visceral Arteries

rupture will be supported by the demonstration
of
biochemical changes in the aneurysm wall induced
by surgical trauma. Kubo et al reported a patient
with extracranial aneurysm presenting as hemorrhage, followed by spontaneous
rupture of a hepatic artery aneurysm [6], although the relationship between
rupture
of the visceral artery
aneurysm and the carotid artery aneurysm resection was not mentioned fully in their report. Rassias
et al reported a patient with an anterior inferior
cerebellar
artery aneurysm and intraabdominal
vessel aneurysm [ 121. This patient suffered two
complications
while
undergoing
hemodynamic
therapy-pulmonary
edema and intraabdominal
hemorrhage-and
they speculated that the latter
was probably the result of the rupture of an aortic
artery aneurysm because of the patient’s precipitous decrease in blood pressure and hemoglobin
concentration.
They further claimed that the increase in transmural pressure induced by the hemodynamic therapy for cerebral vasospasm caused
the rupture of the abdominal vessel aneurysm.
On the other hand, our patient was given thromboxane AZ synthetase inhibitor for cerebral vasospasm in addition
to hemodynamic
therapy.
Thromboxane
A2 synthetase inhibitor is reported
to be effective in the treatment of ischemic symptoms in patients with cerebral vasospasm following
subarachnoid
hemorrhage,
although this drug is
reported to have the side effect of inducing a bleeding tendency during administration
[ 111. In our
case, hemodynamic therapy and the administration
of thromboxane
A2 synthetase inhibitor for symptomatic vasospasm were indicated and the abdominal hemorrhage due to rupture of a rare visceral
aneurysm was impossible to have predicted, yet
our therapy may be more closely associated with
rupture of visceral artery aneurysms than the therapy used by Rassias’s therapy. This complication
involving this form of therapy for vasospasm is
extremely rare, however, this phenomenon
should
emphasize the importance of maintaining an awareness of potential underlying diseases, such as collagen tissue diseases.
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COMMENTARY

Fuse and colleagues describe an interesting patient
with multiple ruptured intracranial and visceral artery aneurysms. The occurrence of intracranial and
intraabdominal
aneurysms in the same individual is
certainly unusual, but not exceedingly rare [l-5]. In
a study from our institution
by Sheps et al [4],
intracranial aneurysms were found in three of 47
patients with splenic artery aneurysms. More recently, the coexistence of abdominal aortic and intracranial aneurysms has been investigated by Norrgard et al [3]. As Fuse and colleagues indicate, an
underlying arteriopathy
is often suspected in patients with systemic aneurysms, but the exact nature of the arteriopathy
is rarely detected.
lt is interesting to note that there appears to be a
propensity for the intraabdominal
aneurysms to
rupture within a few weeks of the craniotomy for
repair of the intracranial aneurysms. Over the last 5
years, we know of at least two patients who suffered