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Oligoastrocytoma presenting with ICH 29

Case Report J. St. Marianna Univ. Vol. 5, pp. 29–32, 2014

Anaplastic Oligoastrocytoma With a Sarcomatous Component Presenting

With Apoplectic Intracerebral Hemorrhage—Case Report

Yohtaro Sakakibara, Masashi Uchida, Yasuyuki Yoshida, and Yuichiro Tanaka

(Received for Publication: April 11, 2014)

AbstractThe authors present a case of apoplectic intracerebral hemorrhage (ICH) caused by an anaplastic oligoas-

trocytoma. A 76-year-old man presented to our hospital with epileptic seizures. A well-demarcated mass was noted at the right temporal lobe upon magnetic resonance imaging and computed tomography scanning. Be-cause the radiological fi ndings of the mass were nonspecifi c, patient follow–up was planned. Two months later, the patient developed a massive ICH at the previously noted area of the right temporal lobe. Right tem-poral craniotomy was performed to evacuate the ICH, and purplish tissue was found at the hematoma cavity rim. This tissue was excised separately for histologic examination. The tumor was diagnosed as an anaplastic oligoastrocytoma with a sarcomatous component (WHO grade III). Postoperatively, the patient was given a combination of radiation and temozolomide therapy. Thereafter, he remained well for 54 months, at which time regrowth of tumor was detected. The patient died of pneumonia 60 months after his initial presentation to our hospital. Oligodendrogliomas and oligodendroglial tumors (OLIGO tumors) most often present with seizures, rarely with apoplectic ICH. Only 1 case report exists of ICH as the initial clinical presentation. Although no explanation has yet been provided as to why apoplectic ICH arising from an OLIGO tumor is such an uncom-mon event, the ICH might be related to the spontaneous rupture of abundant blood vessels with increasing obliteration within the tumor.

Key wordsIntracerebral hemorrhage, Brain tumor, Cerebral apoplexy, Anaplastic oligoastrocytoma

Department of Neurosurgery, St. Marianna University School of Medicine, Kawasaki-shi, Kanagawa, Japan

Introduction

Intracerebral hemorrhage (ICH) caused by a brain tumor is rare1–6). The majority of reported cases of ICH resulting from a brain tumor involve high-grade astrocytoma or metastatic brain tumor3–6). Oli-godendrogliomas and oligodendroglial tumors such as anaplastic oligodendrogliomas and oligoastrocyto-mas (OLIGO tumors) most frequently present with seizures7–9); only rarely do they present with massive ICH2).

We describe our experience with a patient who initially presented with seizures, then later with apo-plectic ICH for which anaplastic oligoastrocytoma was shown to be the cause.

Case Report

History and Presentation. A 76-year-old man was admitted to the emergency room at our institution because of epileptic seizures. Magnetic resonance imaging (MRI) revealed a well-demarcated mass, 2 cm in diameter, located superfi cially at the right middle temporal gyrus. The mass was hypointense on T1-weighted images (Figure 1a), hyperintense on both T2-weighted (Figure 1b) and FLAIR images (Figure 1c), and less enhanced on Gd-DTPA images (Figure 1d). The patient was given prophylactic an-tiepileptics. Because the MRI fi ndings were nonspe-cifi c, follow-up was planned. Two months later, the patient experienced a sudden, severe headache; he

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Sakakibara Y Uchida M et al30

R

R

RR

a b

c d

Figure 1. MRI was performed upon initial presentation. A well-demarcated mass was seen at the right temporal lobe. The

mass is hypointense on the T1-weighted (a) image, hyperintense on both T2-weighted (b) and FLAIR (c) images,

and less enhanced on the Gd-DTPA (d) image.

R

a

R

b

Figure 2. CT scanning was performed upon the second presentation. A massive ICH was seen at the previously noted area of

the right temporal lobe on the plain CT scan (a) and enhanced CT scan (b).

became progressively less responsive and lapsed into unconsciousness. He was brought to our emergency room again. Upon arrival, neurologic examination

showed the patient to be in a state of stupor, with hemiparesis involving the left upper and lower ex-tremities. Computed tomography (CT) scanning

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Oligoastrocytoma presenting with ICH

showed a midline shift and massive ICH at the previ-ously noted area of the right temporal lobe (Figure 2a). Contrast enhanced CT showed no enhancement effect (Figure 2b).Surgery. Right temporal craniotomy was performed to evacuate the hematoma. Tissue on the temporal surface at the rim of the hematoma cavity appeared purplish. This tissue was excised separately for his-tologic examination. The patient regained conscious-ness postoperatively, and the hemiparesis improved quickly.Histologic Examination. Hematoxylin and eosin-stained sections of the surgical specimen revealed cells with bright vacuole and cytoplasm (Figure 3a), and these were positive for glial fi brillary acidic pro-tein (GFAP) (Figure 3b) and oligo-2 (Figure 3c). Also present were spindle-like cells arranged in a storiform pattern. Many bleeding capillaries were

seen in the stroma (Figure 3a). The MIB-1 labeling index was 40% (Figure 3d). The tumor was diag-nosed as an anaplastic oligoastrocytoma with a sar-comatous component (WHO grade III).Postoperative Course. The patient was treated with a combination of focal radiation (60 Gy) and temo-zolomide. After completion of the therapeutic regi-men, the patient remained well for 54 months, at which time regrowth of the residual tumor was de-tected by MRI. His condition deteriorated steadily thereafter, and he died of pneumonia 60 months after his initial presentation to our hospital. Postmortem study was not performed.

Discussion

ICH is an uncommon but recognized initial pre-sentation of brain tumors. According to radiologic and pathologic studies, the incidence of ICH due to

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Figure 3. Histologic fi ndings. Bright vacuoles and cell cytoplasm were seen on hematoxylin and eosin-stained sections of

the surgical specimen; spindle-like cells in a storiform pattern and many bleeding capillaries were also seen in the

tumor stroma (a). The cells were positive for glial fi brillary acidic protein (GFAP) (b) and oligo-2 (c). The MIB-1

labeling index was 40% (d). The tumor was diagnosed as an anaplastic oligoastrocytoma with a sarcomatous com-

ponent (WHO grade III). Magnifi cations: x200 (a and b), x300 (c and d).

a b

c d

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Sakakibara Y Uchida M et al

brain tumor ranges from 0.6% to 14.6%, with an average of 2–3%1) 3) 4–6). Furthermore, the incidence of brain tumor in cases of ICH ranges from 2.5% to 7.2%1) 5). Any type of brain tumor can cause ICH, but the incidence of ICH varies widely among the differ-ent tumor types. Generally, high-grade astrocytoma and metastatic brain tumors are thought to be highly associated with ICH, whereas ICH arising from be-nign brain tumors such as low-grade astrocytoma and meningioma is rare3–5). In cases of OLIGO tumors, microscopically proven but clinically unrecognized intratumoral hemorrhage is not uncommon7) 9). How-ever, OLIGO tumors presenting with macroscopical-ly apparent hemorrhage that seems to have some impact on the clinical course are quite rare. To our knowledge, only 1 case of apoplectic hemorrhage as an initial clinical presentation of OLIGO tumor has been reported in the literature2).

The symptoms of OLIGO tumors do not reliably distinguish them from other types of brain tumor7–9). Seizures are the most common presenting initial symptom, ranging in incidence from 35% to 85% of patients8). Upon CT scanning8), OLIGO tumors ap-pear hypodense or isodense and are enhanced poorly or not at all. Upon MRI, they are typically hypoin-tense on T1-weighted images, hyperintense on T2-weighted images, and less enhanced on Gd-DTPA images. Because both clinical symptoms and radio-logic fi ndings in cases of OLIGO tumors are nonspe-cifi c, patients with oligodendroglial tumor often ex-perience symptoms (usually seizures) for a number of years prior to diagnosis7) 8). The patient in our case was awaiting follow-up after experiencing epileptic seizures for which MRI fi ndings were not specifi c enough to substantiate a proper diagnosis.

Various theories explaining the etiology of ICH arising from brain tumors have been proposed1) 4) 5). The mechanism could be endothelial proliferation followed by vascular obliteration, vessel compression and distortion due to tumor growth, or tumor invasion of the vessel walls. In OLIGO tumors with abundant vessel formation, the vascular structure is composed chiefl y of thin-walled capillaries with an angioma-tous appearance, resulting in endothelial proliferation and necrosis2) 7). In our patient, there were many small bleeding capillaries. We postulate that the spontaneous rupture of these abundant vessels and the increasing vascular obliteration contributed to the pathogenesis of the ICH; however, no explanation has yet been provided as to why apoplectic ICH aris-ing from an OLIGO tumor is such an uncommon

event.

Conclusion

OLIGO tumor should be considered in the dif-ferential diagnosis of unexplained intracerebral hem-orrhage.

Confl ict of Interest Disclosure

The authors report no confl ict of interest con-cerning the materials or methods used in this case or the fi ndings described herein.

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