ee

700 BATJER and PURDY

observed hemosiderin staining of the subarachnoid space
suggestive of subclinical hemorrhage. There are parallels to
this phenomenon in the vascular and thoracic surgical litera-
ture. Aortic dissecting aneurysms have been documented as
enlarging, despite lack of angiographic filling. Presumably.
continued deposition of thrombotic material within the lu-
men of the aneurysm is responsible for its further enlarge-
ment. Indeed. flow into the aneurysm is minimal, below the
resolution of conventional angiography. Given that the nat-
ural history of a totally thrombosed aneurysm is to shrink,
resolve, or calcify: the authors rightly recommend vigilant
follow-up for those lesions found to persist on subsequent

0148-396X/90/2604-0700$02.00/0
NEUROSURGERY
Copyright € 1990 by the Congress of Neurological Surgeons

Neurosurgery, Vol. 26, No. 4

studies. Certainly. those patients who demonstrate sympto-
matic enlargement without angiographic filling should be
considered for operative obliteration to obviate further en-
largement or possibly distal embolization. Conceivably. radi-
olabeled blood products such as platelets. red cells. or fibrin-
ogen may be helpful in documenting flow into an aneurysm.
Turnover of any of these products documented by the appro-
priate scanning techniques might suggest that thrombosis is
not complete.

Steven L. Giannotta
Los Angeles, California

Vol. 26. No. 4. 1990
Printed in U.S.A

Chronic Encapsulated Intracerebral Hematoma Associated with
Cavernous Angioma: A Case Report

Shigeyuki Murakami, M.D., Masahiro Sotsu, M.D., Satoru Morooka, M.D., and Takashi Suzuki, M.D.

Department of Neurosurgery, Aoto Hospital, Jikei University School of Medicine, Tokyo, Japan

The case of a patient with a chronic encapsulated intracerebral hematoma associated with a cavernous angioma is
reported. In spite of a huge space-occupying lesion in the frontal lobe. our patient showed no focal neurological deficit.
The clinical picture suggested a slowly growing brain tumor. Intraoperative findings revealed a liquefied intracerebral
hematoma with a thick capsule. Pathological investigations demonstrated a capsule rich in neovascularity with a
cavernous angioma attached to it. It is presumed that initial bleeding from the cavernous angioma encouraged capsule
formation similar to the membrane of chronic subdural hematomas and that repeated bleeding from the capillaries of
the capsule allowed the expansion of the hematoma. (Neurosurgery 26:700-702, 1990)

Key words: Cavernous angioma, Chronic encapsulated hematoma, Intracerebral hematoma

INTRODUCTION

Intracerebral hematomas that expand gradually like a brain
tumor and produce chronic or slowly progressive neurological
deficits have been reported in the literature. Histologically,
such hematomas are characterized by a thick fibrous capsule
with rich neovascularity. We present a patient with a chronic
encapsulated intracerebral hematoma associated with a cav-
ernous angioma.

CASE REPORT

A 14-year-old boy exhibited increasing headache and vom-
iting over a 2-week period. He was referred to our hospital on
July 6, 1985, because of a presumptive diagnosis of brain
tumor made by a computed tomographic (CT) scan per-
formed at a local hospital. He denied any history of hyperten-
sion, head injury, or coagulopathy.

Physical examination at admission disclosed nothing ab-
normal. Neurological examination revealed no abnormality,
except for blurred margins on the temporal side of the optic
discs bilaterally. All laboratory data including blood coagula-
tion studies were within normal range.

Plain skull x-ray films also disclosed nothing abnormal. A
huge isodense area associated with an eccentric high-density
area adjacent to the inner table of the skull was seen in the

right frontal lobe on the plain CT scan. This was accompanied
by a severe mass effect to the surrounding structures and
perifocal low attenuation (Fig. 1.4). A marginal zone of this
isodense area was partially enhanced by the contrast material
(Fig. 1B). An angiogram of the right common carotid artery
demonstrated a large avascular lesion in the frontal lobe
without any abnormal vessels.

A craniotomy was carried out on July 19, 1985. There was
no adhesion between the dura and the cortex. The cortex
revealed a brownish yellow discoloration. A small amount of
dark brownish fluid similar to the content of chronic subdural
hematomas was aspirated. The thick capsule just beneath the
cortex was incised. The cavity was filled with fluid containing
clots and was irrigated repeatedly with saline. The inside of
the capsule was carefully explored. A small lobulated mass
was found on the wall, which corresponded to the high-density
area shown in the CT scan. There was no abnormal vessel
such as a feeding artery or a draining vein. The capsule was
removed totally, including the mass.

On histopathological examination, the capsule wall was
found to be fibrous connective tissue with rich neovascularity.
Deposits of hemosiderin were also seen in the capsule wall.
These findings were similar to those of the outer membrane
of chronic subdural hematomas (Fig. 24). The specimen
taken from the lobulated mass showed various sized vascular
channels filled with numerous red blood cells. No intervening

April 1990

Fic. 1. 4. plain CT scan revealing a huge isodense area containing
a high-density mass 2 cm in diameter in the right frontal lobe. B, the
margin of the isodense area is partially enhanced.

neural tissue was found. The histological features were con-
sistent with a cavernous angioma (Fig. 2B).

His headache disappeared immediately after surgery, and
the patient was discharged after an uneventful recovery. A
postoperative CT scan revealed a huge low-density area in the
right frontal lobe: however. there was neither a mass effect
nor perifocal low attenuation.

DISCUSSION

The clinical course resulting from cavernous angioma is
usually acute or subacute. and initial symptoms are com-
monly epileptic fits or intracerebral hemorrhage (11). Intra-

CHRONIC ENCAPSULATED INTRACEREBRAL HEMATOMA 701

cerebral hemorrhage may present as a rapidly progressive.
fulminating disease, sudden in onset, with the outcome being
death or profound neurological deficit (6).

We have experienced an unusual case of intracerebral he-
matoma associated with cavernous angioma. Our patient
showed only slowly progressive headache, and there was no
acute episode suggesting intracerebral hemorrhage. Jn addi-
tion, he had no focal neurological deficit such as hemiparesis,
despite the presence of a huge space-occupying lesion in the
right frontal lobe. The clinical picture suggested neoplastic
growth. Intraoperative findings, however, revealed a intracer-
ebral hematoma with a liquefied brownish content similar to
that of chronic subdural hematomas. How do we explain this
unusual clinical course?

Although cavernous angioma is rarely associated with a
large cyst, this association has been described as cystic cavern-
ous angioma (3, 7, 10). These tend to be located in the brain
parenchyma. All of the patients with a cystic cavernous an-
gioma in the cerebral parenchyma reported previously showed
slowly progressive neurological deterioration without acute
episodes (7, 10). The cyst was filled with xanthochromic or
turbid grayish fluid in each reported case. If a mild hemor-
rhage were to occur in the cavity of latent cystic cavernous
angioma, a condition similar to that in the present patient
might be expected.

Recently, patients with intracerebral hematomas who show
stable or slowly progressive neurological deficits have been
reported; these hematomas may be attributed not only to

FiG. 2, 4. photomicrograph of the capsule wall shows fibrous
connective tissue with rich neovascularity (hematoxylin and eosin,
X33). B. the specimen taken from the lobulated mass shows vascular
channels of various sizes, suggesting cavernous angioma (hematoxylin
and eosin. X13).

702 MURAKAMI et al.

hypertension. but also to head injury. anticoagulation therapy.
chemotherapy. or contraceptive medication (12). From the
pathological point of view. a distinct capsule has not been
described in these patients. Hirsh et al. (5) added a new type
of intracerebral hematoma that had a thick, fibrous capsule.
They called this unusual condition chronic encapsulated he-
matoma and suggested that a thick membrane arose from the
fibroblasts related to the abnormal vessels demonstrated in
the thick membrane itself. In the other four reported patients
with thick membranes (1, 2, 8, 9), these abnormal vessels
were not found in spite of a detailed histopathological inves-
tigation; however, neovascularization within the capsule wall
was disclosed in each case. This is similar to the outer mem-
brane of chronic subdural hematomas (2). Even in chronic
subdural hematomas, the cause of chronically expansive
growth is still unknown. Many investigators consider that
recurrent hemorrhages from the outer membrane might play
a major role in this growth (4). Therefore, it is possible to
consider that the initial bleeding encourages capsule forma-
tion with rich neovascularity, and repeated bleeding or exu-
dation from the capillaries of the capsule allows the expansion
of the chronic intracerebral hematoma. The content of the
hematoma may change over time. Takahashi et al. (8) re-
ported a patient with a multilocular encapsulated intracere-
bral hematoma, which had various types of contents including
solid hematoma, liquid hematoma, and xanthochromic fluid
in the different parts of the capsule.

From the above discussion, the lesion in our patient is
considered not to be a cystic cavernous angioma but a chronic
encapsulated intracerebral hematoma because of it had a
thick, fibrous membrane rich in neovascularity. It is con-
cluded that in the chronic encapsulated intracerebral hema-
toma, a repeated hemorrhage from the thick membrane may
play a role in its growth, and a vascular anomaly must be
considered as a cause of initial hemorrhage.

ACKNOWLEDGMENT

The authors extend special thanks to Professor Norio Nak-
amura, Department of Neurosurgery, Jikei University School
of Medicine, for his helpful advice.

Received for publication, August 21, 1989: accepted, final form,
October 19, 1989.

Reprint requests: Shigeyuki Murakami, Department of Neurosur-
gery. Jikei University, School of Medicine, 3-25-8. Nishishinbashi,
Minato-ku. Tokyo, 105, Japan.

REFERENCES

1. Aoki N, Mizuguchi K: Chronic encapsulated intracerebellar he-
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. Aoki N, Mizuguchi K: Expanding intracerebellar hematoma: A
possible clinicopathological entity. Neurosurgery !8:94-96, 1986.

3. Bellotti C. Medina M, Oliveri G, Barrale S, Ettorre F: Cystic

cavernous angiomas of the posterior fossa. Report of three cases.
J Neurosurg 63:797-799, 1985.

4. Bergstram M, Erickson K, Levander B, Svendsen P, Larsson S:
Variation with time of the attenuation values of intracranial
hematomas. J Comput Assist Tomogr 1:57-63, 1977.

. Hirsh LF, Spector HB, Bogdanoff BM: Chronic encapsulated
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6. Paillas JE, Alliez B: Surgical treatment of spontaneous intracer-

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9. Terada T. Okuno T. Moriwaki H. Nakai E, Komai N: Chronic
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10. Vaquero J, Cabezudo JM, Leunda G: Cystic cavernous haeman-
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COMMENTS

Murakami et al. report an interesting case of a chronic
encapsulated intraparenchymal hematoma associated with a
cavernous angioma. Their discussion focuses on the patho-
physiology of this lesion and draws a distinction between
cystic cavernous angioma and chronic encapsulated intracer-
ebral hematoma associated with cavernous angioma. They
conclude that in their patient, the cavernous angioma prob-
ably bled initially, but that the progressive enlargement and
repeated hemorrhage may have been related to the presence
of the vascularized membrane. Although this interpretation
may be correct, I suspect that either repeated bleeding episodes
from the angioma or repeated hemorrhage into a cystic cav-
ernous angioma could produce the same radiographic and
histological situation.

This case highlights the difficulty of managing patients with
non-life-threatening intracerebral hemorrhages without an-
giographically verified pathological findings. In this patient,
the computed tomographic (CT) scan clearly demonstrated a
pathological process, but this is often not the case. Magnetic
resonance imaging (MRI) scans can be extremely helpful in
demonstrating evidence of cavernous angioma, venous an-
gioma, arteriovenous malformation, or neoplasms when acute
CT scans are not helpful. A case can be made for surgically
exploring all hematomas when there is evidence on the MRI
scan of adjacent tissue not felt to be clot. If a nonsurgical
course is followed, repeated angiograms and MRI scans
should be considered after the hematoma has resolved to
identify the occasional arteriovenous malformation rendered
“occult” by a local mass effect.

H. Hunt Batjer
Dallas, Texas

The authors have presented an extremely interesting case,
which is unique, to my knowledge. Although the authors’ case
concerns the growth of an associated hematoma, it is well
known that the cavernous angioma itself is capable of slow
enlargement, perhaps by similar mechanisms. The clinical
spectrum of cavernous angiomas continues to be elucidated.

Theodore Eller
Evanston, Illinois