Neurol Med Chir (Tokyo) 49, 26¿29, 2009

Hemispheric Laminar Necrosis as a Complication of
Traumatic Carotid-Cavernous Sinus Fistula
—Case Report—
Masayasu KATO*,**, Yuka IKEGAME*,**, Izumi TOYODA*, Shinji OGURA*,
Hideomi KITAJIMA**, Shinichi YOSHIMURA**, and Toru IWAMA**
*Advanced Critical Care Center, Gifu University Hospital, Gifu;
**Department of Neurosurgery, Gifu University Graduate School of Medicine, Gifu

Abstract
A 43-year-old woman presented with a very rare case of hemispheric laminar necrosis as a complication
of traumatic carotid-cavernous sinus fistula (CCF). The patient suffered head injury and extensive
burns following a car accident. Oral intubation was performed under sedation. When sedation was discontinued 17 days after injury, the patient demonstrated left hemiparesis. Magnetic resonance imaging
showed laminar necrosis affecting the right cerebral hemisphere. Angiography revealed a right highflow direct CCF. Transarterial embolization of the fistula using a detachable balloon achieved complete
occlusion of the fistula. However, the left hemiparesis persisted following this intervention. Traumatic
CCF may be missed in patients with disturbed consciousness, so clinicians should not overlook possibility of the triad of symptoms of CCF in patients with head injury.
Key words:

carotid-cavernous sinus fistula,

laminar necrosis,

Introduction

head injury

burns involving 40% of her total body surface area.
Oral intubation was performed and the patient was
kept sedated in our hospital intensive care unit.
Several debridement and skin grafting operations
were performed under sedation, during which the
patient's mean blood pressure remained above 80
mmHg and arterial oxygen saturation remained
above 98%. Whole body edema including bilateral
chemosis was noted following extensive intravenous
fluid infusion, but gradually improved except for
right chemosis (Fig. 1A). Sedation was discontinued
17 days after admission, and the patient's consciousness gradually improved. However, the patient then
demonstrated left hemiparesis.
Magnetic resonance (MR) imaging showed abnormal appearance of the cortex and basal ganglia of
the right cerebral hemisphere, dilated bilateral superior ophthalmic veins (SOVs), and abnormal flow
voids in the bilateral cavernous sinuses (Fig. 2). Angiography showed a right high-flow direct CCF with
complete absence of filling of the right internal
carotid artery (ICA) above the fistula (Fig. 3A). The

Carotid-cavernous sinus fistula (CCF) is an abnormal communication between the carotid artery and
the cavernous sinus manifesting as a classical triad
of symptoms consisting of chemosis, proptosis, and
bruit. In general, the prognosis for CCF is good. Lifethreatening complications such as intracerebral
hemorrhage, massive epistaxis, and cerebral ischemia are rare. Here, we report a very rare case of
hemispheric laminar necrosis as a complication of
traumatic CCF.

Case Report
A 43-year-old woman suffered head injury in a car
accident, in addition to extensive burns due to the
car catching fire. On admission to our hospital, the
patient had a Glasgow Coma Scale score of 14 (E3 V5
M6), and demonstrated no motor weakness. Computed tomography (CT) performed 1 hour after the
accident revealed no abnormalities. The patient had

Received December 3, 2007;
Accepted September 5, 2008
Author's present address: M. Kato, M.D., Department of Neurosurgery, Takayama Red Cross Hospital, Takayama, Gifu,
Japan.

26

Laminar Necrosis Caused by CCF

Fig. 1

Photographs of the patient, (A) before treatment for the carotid-cavernous sinus fistula
showing right chemosis and proptosis, and
(B) after treatment showing improved chemosis and proptosis.

Fig. 2

A: Fluid-attenuated inversion recovery magnetic resonance image showing hyperintense areas in the cerebral cortex, caudate
nucleus, and putamen of the right hemisphere. B: T1-weighted magnetic resonance image showing dilated bilateral superior ophthalmic veins (arrows) and abnormal flow voids in the bilateral cavernous
sinuses (arrowheads).

right middle cerebral artery (MCA) was supplied by
cross flow from the contralateral side through the
anterior communicating artery (Fig. 3B). The fistula
drained into the SOV and the basal vein of Rosenthal
(Fig. 3A). We suspected that the laminar necrosis in
the right cerebral hemisphere was due to the CCF.
Transarterial embolization of the fistula using a
detachable balloon resulted in complete occlusion.
Immediately after embolization, both the ICA and
the MCA were completely opacified (Fig. 4). The
patient's chemosis and proptosis improved within a
few days (Fig. 1B). She was discharged with residual
left hemiparesis 2 months after admission.

Neurol Med Chir (Tokyo) 49, January, 2009

27

Fig. 3

A: Lateral right carotid angiogram showing
a right high-flow direct carotid-cavernous
sinus fistula with complete absence of filling of the right internal carotid artery above
the fistula, and venous drainage into the superior ophthalmic vein (arrow) and the basal
vein of Rosenthal (arrowhead). B: Anteroposterior left carotid angiogram showing the right middle cerebral artery supplied
from the contralateral side through the anterior communicating artery.

Fig. 4

Right carotid angiograms showing complete
obliteration of the carotid-cavernous sinus
fistula and the opacified right internal
carotid artery. A: lateral view, B: anteroposterior view.

Discussion
CCFs can be classified angiographically into types A
to D.2) Type A, the most severe type of CCF, is a
direct connection between the ICA and the cavernous sinus. Type A occasionally causes severe complications such as intracerebral hemorrhage, massive epistaxis, decreased visual acuity, and cerebral
ischemia.7) The 3 reported cases of severe cerebral
ischemia caused by traumatic CCF were all type A
(Table 1).9,10,18) Angiography revealed a direct CCF
with total steal, appearing as complete absence of
filling of ICA above the fistula, in two cases.10,18)
CCFs that show complete angiographical ``steal''

M. Kato et al.

28
Table 1

Cases of severe cerebral ischemia caused by carotid-cavernous sinus fistula (CCF)
Barrow's
classification2)

Author (Year)

Cause of
injury

Initial
symptom

Iida et al.
(1995)10)
Iida et al.
(2002)9)

traffic
accident
falling
down

unknown due to
coma
unknown due to
sedation

type A

Wang et al.
(2004)18)

falling
down

aphasia,
hemiparesis

type A

Present case

traffic
accident

unknown due to
sedation

type A

type A

Finding of
angiography
CCF with total
steal
good filling of
the cerebral
artery
CCF with total
steal
CCF with total
steal

Treatment
—
transvenous
coil
embolization
transarterial
balloon
embolization
transarterial
balloon
embolization

Outcome
death
lt hemiparesis
hesitant speech,
mild rt
hemiparesis
severe lt
hemiparesis

Total steal: complete absence of filling of the internal carotid artery above the fistula.

do not always induce cerebral ischemia to the extent
of causing neurological deficits. Seven of 54 patients
with traumatic CCFs had total steal,5) but none of
these seven patients had neurological symptoms.
One case of traumatic CCF with total steal was followed by cerebral infarction in the area of the MCA,
but no collateral flow was present across the anterior communicating artery, so total steal with poor
collateral development probably caused the cerebral
infarction.18) A similar case was followed by a fatal
cerebral infarction.10) Post-mortem examination revealed no thrombus or obstructive change in the
ICA, so that total steal of arterial blood probably
caused the cerebral infarction, similar to complete
obstruction of the ICA.10)
In our case, MR imaging demonstrated cerebral
hemispheric laminar necrosis. Laminar necrosis is
usually seen in anoxic encephalopathy.1) However,
the patient was managed in the intensive care unit,
and arterial oxygen saturation remained above 98%
during her hospitalization. Angiography detected
CCF with total steal, which drained into the basal
vein of Rosenthal. Angiography showed no evidence
of carotid arterial dissection or arterial stenosis after
embolization of the CCF. This unusual venous
drainage may cause deep venous hypertension.6) We
speculate that the reduced blood flow secondary to
the steal phenomenon of the ICA and increased deep
venous pressure due to the CCF led to severe
cerebral hemispheric ischemia, and that this state
continued while the patient sedated, leading to laminar necrosis.
The incidence of traumatic CCFs is only 0.2% to
0.3% among all cases of head or facial injury.10) Detection of traumatic CCF depends on recognizing
the classic triad of symptoms. We did not recognize
the chemosis as a symptom of CCF, and could not
detect cerebral ischemia because our patient was

comatose, so the diagnosis of CCF was delayed and
laminar necrosis occurred. Therefore, other
methods must be applied to identify CCF in
comatose patients. Elevated jugular venous oxygen
saturation (SjO2) established the early diagnosis of a
CCF in a patient with severe head injury.3) Another
case had similar clinical features.12) Therefore, SjO2
monitoring may be useful for detecting CCF in ventilated or sedated patients. Continuous monitoring of
electroencephalography (EEG) and near-infrared
spectroscopy (NIRS) could be safely possible in the
intensive care unit.15,16) EEG monitoring may be useful for indicating outcomes in patients with traumatic brain injury.16) NIRS may be useful for evaluating
brain ischemia.15) The diagnosis of CCF depends
primarily on angiographic findings, but other less
invasive methods, such as MR imaging, CT, and
transcranial Doppler ultrasonography may be used
to screen for CCFs.4,8,10,14) Venous flow signal in the
SOV and cavernous sinus on MR imaging or
abnormal venous flow of the cavernous sinus on MR
angiography are frequently useful for identifying
CCF.8,11) Recently, MR angiography source images
have allowed the diagnosis of CCF without SOV enlargement.8,14) There was a possibility of causing
hemorrhagic infarction by the reperfusion after embolization in this case. The early MR parenchymal
enhancement, which is related to damage to the
blood-brain barrier, may be a good predictor of
symptomatic hemorrhagic infarction in patients
with acute ischemic stroke.17)
The risk of cerebral ischemia is difficult to evaluate based only by the findings of angiography. CCF
with direct shunt (type A) should be immediately
treated following diagnosis.13) Review of angiographic data from 155 patients with CCF identified
various angiographic features associated with risk
of morbidity and mortality, including presence of a

Neurol Med Chir (Tokyo) 49, January, 2009

Laminar Necrosis Caused by CCF
pseudoaneurysm, large varix of the cavernous sinus,
venous drainage to cortical veins, and thrombosis of
venous outflow pathways distant from the fistula.7)
Our present case report suggests that traumatic CCF
may be missed in patients with disturbed consciousness, so clinicians should not overlook possibility of
the triad of symptoms of CCF in patients with head
injury. Continuous monitoring of SjO2, EEG, or
NIRS is necessary for comatose patients. The
presence of total steal detected by angiography is not
always associated with severe cerebral ischemia,
but, given the risk to the patient, early treatment of
CCF with total steal is recommended.

10)

11)

12)

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Address reprint requests to: Masayasu Kato, M.D., Department of Neurosurgery, Takayama Red Cross
Hospital, 3–11 Tenman–cho, Takayama, Gifu
506–8550, Japan.
e-mail: masayasu.kato＠nifty.ne.jp