(Stroke. 1996;27:536-537.)
© 1996 American Heart Association, Inc.
Primitive Malignant Fibrous Histiocytoma of the Neck With Carotid Occlusion and Multiple Cerebral Ischemic Lesions
Marie-Laure Martin-Negrier, MD;
Genevieve Belleannée, MD;
Claude Vital, MD
Jean-Marc Orgogozo, MD
From the Departments of Neuropathology (M.-L.M.-N., G.B., C.V.) and
Neurology (J.-M.O.), CHR Pellegrin, Bordeaux, France.
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Abstract
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Background Emergence of a malignant tumor at the site
of an
operation is a rare event and most often arises in association
with
retained foreign material.
Case Description We describe a patient who 1 year after a
left carotid endarterectomy for typical
atheromatous lesions presented with several
transient ischemic attacks with stepwise worsening of the
deficit and rapid death. A few weeks before, a tumor of the neck had
appeared at the site of the previous
endarterectomy. At postmortem examination, we found
a malignant histiocytofibroma occluding the left carotid artery, with
several recent ischemic foci in the corresponding cerebral
hemisphere without metastasis or tumor emboli.
Conclusions This observation is unusual owing to the
histological type of the neoplasm and to the
circumstance of emergence of the neoplasm.
Key Words: carotid arteries histiocytoma, fibrous foreign bodies sarcoma
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Introduction
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Emergence of a
malignant tumor at the site of an operation is
a rare event and most
often arises in association with foreign
material.
1 2 3 We
report the case of a man who developed a malignant fibrous
histiocytoma
at the site of a previous carotid
endarterectomy performed 1
year before.
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Case Report
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A 73-year-old man was admitted to the hospital for a sudden
right-sided
weakness. One year before admission, he had
presented with several
episodes of right transient brachial
monoparesis, which were
found to be due to a severe left internal
carotid stenosis.
A
thromboendarterectomy of the left proximal
internal and distal
common carotid arteries was performed. The lumen
was 90% stenosed
by an atherosclerotic plaque at macroscopic
examination (no
pathological examination was performed). After the
operation
the patient did well, and 8 months afterward there was no
abnormality
of the artery at Doppler sonography. Two months later
the patient
experienced brief episodes of right transient crural
monoparesis
and then the symptoms that led to hospitalization. On
admission
the patient presented with a paresis of the right
lower limb,
followed quickly by a partial recovery; however, 2 days
later
he had a new attack of severe right-sided hemiparesis.
Doppler
sonography showed a left internal carotid occlusion by an
anechogenic
material and an atheromatous plaque at the
bifurcation. A cranial
CT scan showed no focal hypodensity. During
hospitalization
the patient complained of a pain in the left side of
the neck
during swallowing. On physical examination no mass was found,
and
a diagnosis of postoperative fibrosis was made. Analgesics were
prescribed,
and the pain decreased. A few weeks later a left lateral
neck
mass was discovered after a worsening of the local pain. On
physical
examination the patient presented with a marked right
hemiparesis.
Doppler sonography still showed an occlusion of the
left primitive
carotid and internal carotid arteries. A cranial CT scan
showed
a small left cortical infarct. Ultrasonography of the neck mass
showed
a heterogeneous, fixed, hypoechogenic and
hyperechogenic mass
measuring 30 mm in diameter located anterior to the
sternocleidomastoideus,
suggesting a lymph node enlargement. Before the
decision to
operate was made the physical state of the patient
worsened,
and he died 1 week later. An autopsy restricted to the area
of
the neck mass and to the brain was performed.
At postmortem examination a firm, solitary, multilobated,
pseudoencapsulated tumor 10x6x4 cm was found in the area of the
left
carotid bifurcation. On cut sections the mass was surrounding the
carotid artery, and surgical threads (from the previous carotid
endarterectomy) were found within the tumor.
Microscopic examination revealed a relatively well-circumscribed
tumor consisting of clump spindle cells arranged in a storiform pattern
and in giant multinucleated cells. The mitotic activity was high, with
17 mitoses per 10 high-power fields. The stroma consisted of
collagen fibrils, occasionally with a marked collagenization or focal
myxoid change. The vasculature consisted of small vessels with areas of
hemangiopericytoid pattern. Large areas
of necrosis were widespread in the tumor. This malignant proliferation
was surrounding, infiltrating, and occluding the carotid artery. At
immunochemistry, the tumor was negative for the epithelial markers
(CYTO K-, EMA-) and for
protein S 100 (a marker of melanoma and Schwann cell tumor), positive
for vimentin (mesenchymal marker), and negative for desmin and HHF35
(muscular markers). The diagnosis of storiform pleomorphic type grade 3
malignant fibrous histiocytoma was made. Multiple foreign body
granulomatous reactions were found in the proximity of the tumor around
the surgical threads of the previous
endarterectomy.
Macroscopic examination of the brain showed a few small nonocclusive
atheromatous deposits in the vessels of the circle of
Willis. The coronal sections revealed several whitish areas in the
subcortical white matter of the left cerebral hemisphere. At the
microscopic level, recent ischemic lesions were observed in the
left temporal, frontal, and parietal cortex and in the left hemispheric
white matter. No metastasis or emboli of the malignant fibrous
histiocytoma were seen in the brain.
The diagnosis of left multiple ischemic brain foci secondary to
left carotid occlusion by the malignant fibrous histiocytoma was
established.
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Discussion
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Malignant fibrous histiocytoma is a soft-tissue tumor of late
adult
life; the majority of cases occur in people older than 50
years.
4 5 The common locations are the extremities,
followed by the
retroperitoneum. The neck is a very unusual
site
4 5 ; only one
other case of malignant fibrous
histiocytoma of the neck has
been reported, to our
knowledge.
6
The development of a malignant tumor at the site of an operation is
very unusual. Although it seems unlikely, the first possibility is that
the tumor was present at the time of the endartectomy, in the
absence of pathological examination to rule out this hypothesis. The
second possibility, which we find more plausible, is the emergence of
the malignant tumor on the scar of the
endarterectomy. Indeed, a few cases of sarcoma
arising at the site of a previous operation have been
reported.1 2 3 In most of these cases,
the sarcoma was
associated with foreign material and arose after a very variable
latency period.1 2 3 The foreign material
may have different
physicochemical characteristics and mainly is associated with
prostheses for joint replacement and vascular
grafts.1 2 3
In our case surgical threads were found within and in the vicinity of
the malignant proliferation and therefore could have induced
tumorigenesis. Studies in animals have shown that the physical presence
and not the chemical components of the implant of foreign bodies may be
responsible for tumorigenesis.7 The most critical factor
in the induction of these sarcomas is the formation of a fibrous
capsule around the foreign body.7 In our case the
persistence of a foreign body granulomatous reaction and the presence
of large extensive fibrosis areas in the tumor seem to be in agreement
with this possibility. Since the number of carotid endarterectomies is
increasing after the positive results of several prevention trials, the
possibility of occasional induction of sarcomas at the site of the
operation should be kept in mind.

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Figure 1. Carotid artery with sarcomatous proliferation
outside the carotid wall and in the lumen (arrows). Bar=100
µm.
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Figure 2. Detail of the sarcomatous proliferation with
numerous giant multinucleated cells. Bar=20 µm.
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Acknowledgments
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We are grateful to Professor Boris Sandler for the translation
of
the Russian article
6 and to Ray Cooke for linguistic
help.
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Footnotes
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Reprint requests to M.-L. Martin-Negrier, MD, Service
d'Anatomopathologie,
CHR Pellegrin, F-33076 Bordeaux, Cedex, France.
Received August 21, 1995;
revision received November 6, 1995;
accepted November 21, 1995.
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References
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Enzinger FM, Weiss SW. Malignant
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