DOI: 10.5137/1019-5149.JTN.11004-14.1
Received: 19.03.2014 / Accepted: 09.05.2014

Case Report

Spontaneous Intracerebral Hematoma in Low-Grade Glioma
After 14 Years of Follow-Up
Miloš JokovIć1,2, Vojislav BogosavljevIć1,2, Igor NIkolIć1,2, Nemanja JovanovIć1
Clinical Center of Serbia, Clinic for Neurosurgery, Belgrade, Serbia
University of Belgrade, School of Medicine, Belgrade, Serbia

1
2

ABSTRACT
We are reporting the case of a 53-year old woman presenting to our hospital with a hemorrhagic low-grade glioma (LGG). She
was admitted to a nearby general hospital where she had presented with aphasia, right hemiplegia and change of mental status.
Computer tomography (CT) images showed a left temporo-parietal hemorrhage with mass effect. She was transferred to our
hospital neuro-intensive care unit where emergency craniotomy was performed. A tumor with hematoma was removed and further
histopathology analysis revealed tumor progression.
We reviewed the literature reporting cases of central nervous system tumors hemorrhage and found that these types of events
are exquisitely rare in adults with LGG. However these events are possible, suggesting that it should be included in the differential
diagnosis of any patient presenting with intracranial hemorrhage. This case raises questions regarding the benefit of early versus
late intervention for patients known to have LGG.
Keywords: Intracerebral hematoma, Low-grade glioma, Surgery

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Introduction

Although intratumoral hemorrhages can occur with any brain
tumors, it is a very rare occurrence with low-grade glioma
(LGG). In general, brain tumors are associated with intracranial
hemorrhage 1-10% of the time, with an average incidence
of 3.3%. In clinical series, gross hemorrhage from gliomas
was seen in 1.6-10% , while the incidence of hemorrhage for
all cerebral neoplasms ranged from 0.7 to 6%. Intracranial
hemorrhage is commonly seen in children (1,2).
We reviewed the literature of cases with bleeding in central
nervous system tumors and reported our case of hemorrhagic
low-grade glioma in a 53-year-old woman presenting with
right hemiplegia, aphasia and coma. Computer tomography
(CT) images showed a left temporoparietal hemorrhage with
mass effect.

Corresponding author: Vosjislav BogosavljevIć
E-mail: neurobog1171@gmail.com

452 | Turk Neurosurg 26(3):452-455, 2016

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Case report

The patient was a 53-year-old woman presenting with acute
onset right hemiplegia and coma. CT scan showed a tumor in
the temporo-parietal region of the brain in the left dominant
hemisphere (in the insular region). Before this admission,
we had followed this patient for thirteen years for a tumor in
the temporo-parietal region. The initial disease presentation
was a Jackson seizure. A CT with and without contrast was
performed and showed the existence of a hypodense lesion in
the relevant region (Figure 1). This lesion was not associated
with any mass effect. Based on CT characteristics, the lesion
was felt to be most consistent with a LGG. Taking into account
the localization and the fact that we were dealing with a
tumor in the dominant hemisphere, the decision was taken to
postpone surgery and follow-up. The patient was prescribed
antiepileptic treatment. The plan for regular monitoring meant
primarily to perform diagnostic CT and/or magnetic resonance
imaging (MRI) every 6 months for the first year, and then each

Joković M. et al: Low-Grade Glioma

Figure 1: First CT from 1999.

A

b

c

Figure 2: A) MR spectroscopy in 2007.
B) T2 Flair, C) T1-W MRI in 2011 show
left insular tumor.

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Joković M. et al: Low-Grade Glioma

year. Intraoperative monitoring was not available in the country
at that time. A year later, the patient had a second seizure but
this had no clinical consequences (normal neurological status,
no subjective symptoms). A control CT with and without
intravenous contrast did not show tumor progression. MRI
was performed and confirmed the presumed diagnosis of
LGG (Figure 2A-C). MR spectroscopy revealed reduction of
NAA and elevation of Cho, myoinositol and lipid peak (Figure
2A).
The patient was started on a higher dose of antiepileptic drugs
and she did well, with no seizure activity and no symptoms
until October 2013, when she developed a sudden and
severe headache with weakness of the right extremities and
mild dysarthria after waking up in the morning. By the time
of her examination in the nearest general hospital, she had
developed complete right hemiplegia and aphasia. A CT scan
was performed, and showed an intracerebral hematoma on

the left side of the temporo-parietal part of the brain, in the
same region where the LGG had previously been identified
(Figure 3).
She was transferred to the intensive care unit in our hospital.
During transport, and shortly before admission to our hospital,
there was a disturbance of consciousness leading to coma
requiring artificial ventilation support. The GCS score was 7
with right hemiplegia and a discreetly asymmetrically enlarged
left pupil. After preoperative preparations, emergency surgery
was performed.
An intraoperative tumor was found with markedly proliferating
blood vessels and with the presence of spontaneous intratumoral and peritumoral hematoma. The tumor with hematoma
was removed. She was extubated on the first postoperative
day and postoperative recovery was gradual. She recovered
consciousness and the neurological deficit gradually decreased over the next couple of days. On the third postoperative day, the right hemiplegia changed to a barely noticeable hemiparesis and she started recovering the capacity of
pronouncing certain words. She was started on early physical
treatment. A control CT scan on the first post-operative day
showed a small blood collection and no signs of residual tumor.
Histological examinations showed an anaplastic astrocytoma
WHO grade III with the classic image of fibrillar astrocytes,
without an oligodendroglial component and calcification
The tissue was well vascularized and some blood vessels
showed the existence of endothelial hyperplasia (Figure 4A,B).
Immuno-histopathological analysis showed GFAP + + +, CD57
+ + + + + p53, WT-1 and Synaptophysin + + + +, while S100,
vimentin, CD10, RCC, TTF1, CK and AE1/AE3 were negative.
Ki67 was 16.86%.

Figure 3:
Preoperative CT
in 2013.

A

Based on the above history and histopthological findings, the
neurooncological council made the decision to administer
postoperative radiotherapy with concomitant chemotherapy.
Four months later the patient was communicative and independently mobile with full recovery of the neurological deficit

B

Figure 4: Histopathological findings; A) tumor with bleeding (HE x40); B) tumor with bleeding and neovascularisation (HE x100).

454 | Turk Neurosurg 26(3):452-455, 2016

Joković M. et al: Low-Grade Glioma

findings in our patient indicate, endothelial hyperplasia was
seen in a number of intratumoral blood vessels. Abnormal and
weak blood vessels are mainly associated with higher grade
gliomas. This fits with the findings seen in our patient in whom
dedifferentiation and progression from a low-grade glioma in
an anaplastic form (WHO gr III) appears to have occurred . It
is now generally accepted that the main cause of mortality
in these tumors is their dedifferentiation to a higher degree
of malignancy (7,8,9). Clinical studies of miR-544 in tumor
tissue have shown that expression decreased significantly
in secondary anaplastic astrocytoma, and in cases of
spontaneous progression in malignant forms (6).

Figure 5:
Postoperative
CT four months
after operation.

(right hemiparesis). Repeated CT scans showed an intraparenchymal defect at the site of the tumor apoplexy, and no
residual tumor (Figure 5).
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Discussion

At the time of her initial presentation, it was felt that the patient
had a LGG occupying part of her dominant hemisphere. Due
to the tumor characteristics, as well as the fact that our patient
had no neurological deficits, it was felt that the risk/benefit
ratio associated with surgery was too high, and it was decided
that a watch and wait approach was the most reasonable. The
patient was followed for thirteen years with regular follow-up
CT / MRI, which showed no signs of tumor progression and
no changes in enhancement. Before the bleeding episodes,
no clinical or radiographical signs of disease progression
were seen. Intracerebral hematoma is an unusual clinical
presentation for low-grade gliomas and it has been described
in a small number of cases in adults (3,4,5). Bleeding in glioma
is described mainly in tumors that have malignant properties
(1).
The reasons for the emergence of intratumoral bleeding in
low-grade gliomas are unclear. Probable causes of bleeding
are abnormal formation of intratumoral blood vessels and
endothelial proliferation within them. As the histopathological

This case illustrates that the decision to delay surgery for
patients with LGG should perhaps be changed, and that it
may in fact be beneficial for patient to have surgery sooner
rather than later.
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REFERENCES

1. Della Puppa A, Zustovich F, Gardiman M, Manara R, Cecchin
D, Scienza R: Hemorrhagic presentation of low-grade glioma in
adults. Acta Neurochir (Wien) 149:1151–1155, 2007
2. Garg A, Chugh M, Gaikwad SB, Chandra SP, Gupta V, Mishra
NK, Sharma MC: Juvenile pilocytic astrocytoma presenting
with subarachnoid hemorrhage. Case report and review of the
literature. J Neurosurg 100 Suppl 5:525–529, 2004
3. Gottfried ON, Fults DW, Townsend JJ, Couldwell WT: Spontaneous
hemorrhage associated with a pilomyxoid astrocytoma: Case
report. J Neurosurg 99: 416-420, 2003
4. Kondziolka D, Bernstein M, Resch L, Tator CH, Fleming JF,
Vanderlinden RG, Schutz H: Significance of hemorrhage into brain
tumor: clinicopathological study. J Neurosurg 67:852–857, 1987
5. Licata B, Turazzi S: Bleeding cerebral neoplasm’s with
symptomatic hematoma. J Neurosurg Sci 47:201–210, 2003
6. Ma R, Zhang G, Wang H, Lv H, Fang F, Kang X: Down regulation
of miR-544 in tissue, but not in serum, is a novel biomarker of
malignant transformation in glioma. Oncol Lett 4:1321-1324, 2012
7. Mineo JF, Bordron A, Baroncini M, Maurage CA, Ramirez C,
Siminski RM, Berthou C, Dam Hieu P: Low HER2-expressing
glioblastomas are more often secondary to anaplastic transformation of low-grade glioma. J Neurooncol 85:281-287, 2007
8. Radulović D: Natural history of supratentorial low-grade
astrocytoma: Case report. Srp Arh Celok Lek 134:537-540, 2006
9. Tungaria A, Sahu RN, Kumar R, Srivastava A: Intratumoral
hemorrhage in brainstem low-grade glioma. Neurol India 60:
243-245, 2012

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