364

SHORT REPORT

A case of gliosarcoma appearing as ischaemic stroke
S Züchner, W Kawohl, B Sellhaus, M Mull, L Mayfrank, C M Kosinski
.............................................................................................................................

J Neurol Neurosurg Psychiatry 2003;74:364–366

Objectives: Ischaemic stroke attributable to malignant
brain tumour is a rarely reported phenomenon and even
various imaging techniques including angiography do not
necessarily lead to an accurate diagnosis.
Case description: A 46 year old, previously healthy man
developed apoplectic symptoms with slight right sided
hemiparesis and global aphasia. The computed tomography (CT) scan showed lesions of the left temporal lobe and
the paraventricular white matter suggestive of left middle
cerebral artery (MCA) infarction. Carotid angiography
demonstrated compression of the M1 segment of the MCA
and occlusion of temporal MCA. The patient initially
refused magnetic resonance imaging (MRI) because of
claustrophobia. Because of fluctuating symptoms and successive worsening of the condition over weeks an MRI
scan was conducted under general anaesthesia. Beneath
temporal, opercular, and subcortical infarctions it revealed
a left temporal tumour. A tumour biopsy disclosed a
gliosarcoma (WHO grade IV). Microscopical examination
of the surgical specimen demonstrated invasion of tumour
cells into the wall of a greater pre-existing blood vessel.
Conclusions: Malignant brain tumours may cause ischaemic infarction. This is a rare but important differential diagnosis for the origin of strokes. The authors describe the first
case with infiltration of intracranial blood vessels by
tumour cells of a gliosarcoma.

I

schaemic strokes are most commonly caused by thrombotic
obstructions of extracranial or intracranial vessels on the
basis of atherosclerotic or non-atherosclerotic vasculopathies and “cardiac” or vascular cerebral embolism. Acquired or
genetically determined coagulopathies and haematological
diseases are rare causes. In contrast, cerebral neoplasms are
known to induce apoplectic onset of neurological symptoms
by intracranial haemorrhage.1 2 In very rare cases malignant
tumours have provoke a sudden onset of neurological
symptoms attributable to ischaemic stroke.3 Most commonly
meningeomas have been known to cause stenosis or occlusion
of intracranial vessels by vascular compression or
encasement.4 5 Aoki et al describe a case of a malignant glioma
causing a cerebral infarction by dissection of the middle
cerebral artery caused by invasion of the vessel wall.6 Herman
et al observed leptomeningeal dissemination of a malignant
glioma, which mimicked cerebral vasculitis.7 Here we report a
case with cerebral infarction as the apoplectic clinical
manifestation of a gliosarcoma with atypical angiographic and
neuropathological features.

CASE REPORT
A 46 year old man without significant medical history was
admitted with acute onset of a slight right sided weakness and
global aphasia. The symptoms were fluctuating. The neurological examination disclosed no additional deficits. Computed tomography (CT) scans demonstrated lesions of the left
temporal lobe and the paraventricular white matter, which

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were initially interpreted as middle cerebral artery (MCA)
infarction (fig 1A, 1B). Because of claustrophobia the patient
refused magnetic resonance imaging (MRI). Doppler sonography of the extracranial and intracranial arteries revealed no
stenosis, occlusion, or dissection. Transoesophageal echocardiography, 24 hour Holter monitoring, and blood screens
for coagulopathies were completely normal. The symptoms
subsided within 10 days to a mild mixed aphasia and the
patient was dismissed with clopidogrel. Five days later the
aphasia worsened again and the patient developed a right
sided hemiparesis. A CT follow up scan disclosed a progressive
lesion extending from the left temporal region to the left
operculum and the left paraventricular white matter. Transcranial duplex sonography of the cerebral arteries now revealed
narrowing of the trunk of the left MCA. Cerebral angiography
showed narrowing of the sphenoidal, insular and opercular
parts of the left MCA and occlusion of the left temporal arteries at the MCA bifurcation (fig 1C). No pathological vessels
indicating a tumour were observed. Repeated cerebrospinal
fluid examinations showed mild lymphocytic pleiocytosis (15
cells/µl) but no specific germ or tumour cells. Despite negative
results of serological examinations an isolated vasculitis of the
CNS seemed still possible and the patient received subsequently on a trial basis a treatment with corticosteroids leading to a temporary improvement of the condition over six
weeks. Because of fluctuating symptoms and clinical deterioration MRI was conducted under general anaesthesia seven
weeks after readmission. In addition to the known infarctions
it showed a hyperintense temporal lesion with gadolinium
enhancement, suggesting a neoplasm (fig 1D). The patient
underwent subtotal removal of the tumour via right frontotemporal craniotomy. The postoperative course was uncomplicated. Neuropathological examination revealed a malignant
brain tumour presenting a biphasic tissue pattern with
gliomatous and mesenchymal components. Gliomatous areas
showed typical features of a glioblastoma with remarkable
nuclear atypia, ischaemic necroses, increased mitotic index,
and glial fibrillary acidic protein (GFAP) expression. The mesenchymal component consisted of areas with densely packed
long bundles of spindle cells in a storiform pattern. Immunohistochemically the cells of this section expressed vimentin
and were surrounded by a dense network of reticulin fibres as
typical features of sarcomatous cells. Within the gliomatous
section GFAP positive tumour cells infiltrated the whole
adventitia and muscle layer up to the intima of one large blood
vessel (fig 2). Whether the vessel is venous or arterial is difficult to determine because of the deep infiltration of the
tumour cells into the wall. Although the vessel is related to a
necrotic area, a complete occlusion was not seen. The tumour
was classified as a gliosarcoma (WHO grade IV). Additionally,
the patient received 60 Gy of radiation therapy. Eight months
later he died. The relatives refused a postmortem examination.

.............................................................
Abbreviations: MRI, magnetic resonance imaging; CT, computed
tomography; MCA, middle cerebral artery

Gliosarcoma appearing as stroke

365
Figure 1 Neuroradiological
findings. (A, B) Cranial CT scans two
days after onset of symptoms show
small hypodense lesions of the left
centrum semiovale (arrows). (C)
Posteroanterior view of a left carotid
arteriogram three weeks after onset of
symptoms revealed stenosis and
occlusion of the left temporal artery
(arrow). (D) MRI scan six weeks after
onset of symptoms reveals a
hyperintense lesion of the left
temporal lobe in the T1 weighted
image with contrast enhancement,
(arrow) right sided developmental
venous anomaly.

Figure 2 Tumour specimen immunohistochemically stained with
antibody against GFAP. The large vessel (inner diameter 0.16 mm)
is infiltrated by GFAP positive gliomatous tumour cells, but not
occluded. Pleomorphic tumour cells were apparent in the adventitia
and media of the vessel wall, (*) necrotic area.

DISCUSSION
The sudden onset of clinical symptoms and the hypodense
lesions in the CT scans (fig 1A, 1B) were initially considered as
a “classic” ischaemic stroke. The recurrent infarctions and the
mild lymphocytosis of the cerebrospinal fluid, the narrowing
of MCA branches in the cerebral angiography (fig 1C) as well
as the clinical stabilisation under corticosteroid treatment over
six weeks were suggestive of a cerebral vasculitis. The lack of

typical signs made it difficult to diagnose a malignant tumour.
Moreover, the claustrophobia of the patient made it impossible
to conduct MRI, initially.
It has occasionally been observed that gliomatous cells are
able to invade the arterial wall and may induce weakening of
the wall leading to arterial dilatation or rupture.8 9 However,
gliosarcoma is an uncommon malignant brain tumour with
mixed glial and mesenchymal elements. The neuropathological examination of the presented case demonstrated the invasion of the wall of a major blood vessel by partially necrotic
gliomatous tumour cells (fig 2). Tumours usually develop more
irregular vessels. Thus, we argue that this vessel is not a
tumour neovascularisation but a pre-existing blood vessel. The
authors propose a haemodynamically induced low flow
infarction attributable to compression and progressive occlusion of MCA branches by tumour cells.10 Although the intima
of the observed vessel was intact in other sections a breach of
the intima may induce vascular thrombosis and arterioarterial embolism. This is the first reported stroke resulting
from invasion of the wall of intracranial arteries by gliomatous
cells of a gliosarcoma. The reason why a small subgroup of
malignant gliomas becomes primarily symptomatic by infiltrating the walls of the blood vessels instead of the extending
volume remains to be explained.
In conclusion, tumour infiltration of major intracranial
blood vessels by malignant gliosarcoma can cause ischaemic
infarction. In early stages of the disease it can be a difficult but
important differential diagnosis for recurrent cerebral infarction.

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366

Züchner, Kawohl, Sellhaus, et al

.....................

Authors’ affiliations
S Züchner, C M Kosinski, Department of Neurology, Aachen University
Hospital, Aachen, Germany
S Züchner, B Sellhaus, Department of Neuropathology, Aachen
University Hospital
W Kawohl, Department of Psychiatry, Aachen University Hospital
M Mull, Department of Neuroradiology, Aachen University Hospital
L Mayfrank, Department of Neurosurgery, Aachen University Hospital
Competing interests: none declared.
Correspondence to: Dr S Züchner, Department of Neuropathology,
University Hospital of Aachen, Pauwelsstrasse 30, 52074 Aachen,
Germany; stephan.zuechner@post.rwth-aachen.de
Received 5 June 2002
In revised form 23 September 2002
Accepted 23 October 2002

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