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Suprasellar Endodermal Sinus Tumor Presenting with Tonic-Clonic Seizure - an Autopsy Case Report

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Suprasellar Endodermal Sinus Tumor Presenting with Tonic-Clonic Seizure - an Autopsy Case Report 대한병리학회지: 제 36 권제6 호 2002 The Korean Journal of Pathology. 2002; 36: 433-9 Suprasellar Endodermal Sinus Tumor Presenting with Tonic-Clonic Seizure - An Autopsy Case Report - Min Jin Lee∙Heasoo Koo We report the clinical course and autopsy findings of a 19-year-old girl with endodermal sinus Woon Sup Han∙Sun Hee Sung tumor involving the thalamus, hypothalamus, and basal ganglia. The patient initially had tonic- Yong-Jae Kim1∙Hye Young Choi2 clonic seizures with abnormal signal involving the right hippocampus, amygdala, basal gan- glia, putamen, and dentate gyrus. The signal intensity of the posterior pituitary on T1-weight- ed images was decreased at the time of admission, which was not associated with clinical symp- Departments of Pathology, 1Neurology, and 2Radiology, Ewha Womans toms of diabetes insipidus (DI). A huge tumor mass as well as central DI developed within 10 University, College of Medicine months. The postmortem examination showed gliosis with calcification involving the right basal Seoul, Korea ganglia, internal capsule, and white matter, in addition to a tumor mass involving the thala- mus, hypothalamus, and basal ganglia. Dissemination of tumor cells in the leptomeninges and the gliotic area and hydrocephalus were also noted. Received : July 25, 2002 Accepted : October 4, 2002 Corresponding Author Heasoo Koo, M.D. Department of Pathology, College of Medicine, Ewha Womans University, 911-1 Mok-dong, Yangcheon-gu, Seoul 158-056, Korea Tel: 02-650-5730 Fax: 02-653-8891 E-mail: [email protected] Key Words : Brain Neoplasms-Endodermal Sinus Tumor-Epilepsy, Tonic-Clonic Intracranial endodermal sinus tumor (EST) is a rare type of tumor cells as well as central DI developed within 10 months. germ cell tumor (GCT) and it comprises 7-10% of primary The postmortem examination showed gliosis with infiltrating intracranial GCTs.1-11 EST occurring in the sellar region is less tumor cells and calcification involving the right basal ganglia, common than the one occurring in pineal region, which is sim- internal capsule, and white matter, in addition to a tumor mass ilar to other histological types of GCT. Sellar or suprasellar GCT, involving the thalamus, hypothalamus, and basal ganglia. including EST, manifested visual field defect as well as symptoms and signs of distrupted hypothalamo-hypophyseal axis, such as diabetes insipidus (DI), pituitary failure, and precocious puber- CASE REPORT ty.1-7,9,11-18 Seizures or involuntary movements were rarely report- ed in cases of intracranial GCT.1-3,6,9 The magnetic resonance (MR) The patient came to the hospital with suddenly developed findings of isolated pituitary stalk thickening and/or pituitary generalized tonic-clonic seizures on November 30, 1997. She gland enlargement or absence of hyperintense signal generated was found in an unconscious state and brought to the emergen- by a normal neurohypophysis on a T1-weighted image were cy room. The patient had a past history consisting of intermit- well documented in GCT involving the hypothalamo-pituitary tent headaches. The brain computed tomography (CT) showed area.3,6,7,12,13,15-18 We report a case of suprasellar EST initially pre- an abnormal signal involving the right hippocampus, amygdala, sented with tonic-clonic seizures and an abnormally high signal basal ganglia, putamen, and dentate gyrus (Fig. 1). A topograph- intensity involving the thalamus, hypothalamus, and basal gan- ic sagittal T1-weighted image showed a slightly decreased sig- glia. A huge tumor mass with leptomeningeal dissemination of nal intensity at the posterior lobe of the pituitary gland com- 433 434 Min Jin Lee∙Heasoo Koo∙Woon Sup Han, et al. A B Fig. 1. The brain computed tomography (A) shows high density at the right basal ganglia (arrow) and the T1-weighted image of the brain (B) reveals high signal intensity at the right basal ganglia, globus pallidus, and posterior putamen (arrow). A B Fig. 2. The T1-weighted sagittal image (A), taken during first admission, shows a slightly decreased high signal intensity (arrow) at the pos- terior lobe of the pituitary gland compared to that of the anterior lobe. The sagittal T1-weighted image (B), taken on October 8th, 1998, reveals a similar signal intensity (arrow) on the posterior pituitary gland compared to that of the anterior lobe, in addition to a large tumor mass with low signal intensity at the suprasellar area. pared to that of the anterior lobe (Fig. 2A). She was treated with but did not show any neurological symptoms or signs. She was Orfil and Dilantin during regular follow-up visits until Septem- admitted to the hospital on October 2nd, 1998 with a chief com- ber 26th, 1998 because the was thought to have epilepsy. Serum plaint of abrupt mental status change. The T1-weighted axial or cerebrospinal fluid (CSF) examinations for tumor markers or image of the brain showed a large and well-defined oval shaped hormone levels or the radiological follow-ups were not performed isoechoic mass at the suprasellar area, which had amorphous high during the period. She complained of intermittent headaches, signal hemorrhage within the mass (Fig. 3A). The T2-weight- Suprasellar Endodermal Sinus Tumor 435 A B Fig. 3. The T1-weighted axial image of brain (A) shows a large and well defined oval shaped isoechoic mass at the suprasellar area, which has amorphous high signal hemorrhage within the mass. The T2-weighted axial image of the brain (B) shows an inhomogeneous high sig- nal mass at the third ventricle area with mild hydrocephalus. which was treated with pitressin. Levels of serum alpha fetopro- tein (AFP), beta-human chorionic gonadotropin ( -HCG), and carcinoembryonic antigen (CEA) were 13435.5 ng/mL (normal: <12.5 ng/mL), 23.28 mIU/mL (normal: <5.0 mIU/mL), and 2.0 ng/mL (normal: <5.0 ng/mL), respectively. Levels of CSF AFP and CEA were 72464.7 ng/mL and 0.6 ng/mL, respective- ly, and cell count revealed 20 red blood cells and 40 white blood cells. Emergency radiotherapy and hormonal therapy were rec- ommended but refused by patrons. The patient died on October 20th, 1998. A limited autopsy was performed on brain. The brain weighed 1,556 g and showed prominent parahippocampal gyri with lon- Fig. 4. The coronal section of the brain shows a large hemorrhag- gitudinal grooves. The leptomeninges revealed multifocal pale ic and necrotic tumor mass occupying the thalamus, hypothala- yellowish thickening with entrapped cranial nerves over the base mus, and basal ganglia, which infiltrates into the subarachnoid of the brain. The markedly hemorrhagic and necrotic 5×4×3 space. The lateral ventricle is markedly dilated with interstitial edema in surrounding white matter. Compared with left side, the cm sized tumor mass occupied large areas of the thalamus, hypo- right putamen, globus pallidus, internal capsule, and periventric- thalamus, and basal ganglia with subarachnoid dissemination ular white matter reveal a poorly defined yellowish brown atroph- (Fig. 4). The lateral ventricles were markedly dilated and sur- ic area (arrows). rounding tissues showed edematous changes. The right putamen, ed axial image of the brain showed an inhomogeneous high sig- globus pallidus, and internal capsule, as well as the white mat- nal mass at the third ventricle area with mild hydrocephalus ter, revealed a poorly defined pale brownish atrophic area. Micro- (Fig. 3B). After contrast enhancement, the mass, except focal scopic examination of the tumor mass showed a reticular pattern necrotic areas, was almost homogeneously enhanced. The sagit- of endodermal sinus tumor, consisting of a loose meshwork of tal T1-weighted image, taken on October 8th, 1998, revealed a communicating spaces lined by poorly differentiated epithelial signal intensity at the posterior pituitary gland that was similar cells and frequently noted Schiller-Duval bodies (Fig. 5A). There to that at the anterior pituitary gland (Fig. 2B). The patient devel- were also sheets and compact nests of large cells with irregular oped symptoms of central DI with hypernatremia and polyuria, vesicular large nuclei, prominent nucleoli, and eosinophilic or 436 Min Jin Lee∙Heasoo Koo∙Woon Sup Han, et al. A B C D Fig. 5. Microscopic examination of the tumor (A) shows a reticular pattern of endodermal sinus tumor, consisting of a loose meshwork of communicating spaces lined by poorly differentiated epithelial cells with Schiller-Duval bodies. The PAS stain (B) reveals many bright eosinophilic PAS-positive intracytoplasmic and extracellular hyaline bodies (arrows). The immunohistochemical stains show positive reac- tion to alpha fetoprotein (C) and strong positive reaction to CAM5.2 (D). clear cytoplasms. Mitotic figures and bright eosinophilic PAS Glostrup, Denmark), -HCG (1:50, Zymed), and CD30 (1:30, positive intracytoplasmic and extracellular hyaline globules were Dako) antibodies (Fig. 5C & D). There were multifocal nests of numerous (Fig. 5B). The tumor cells showed strong positive tumor cells along the leptomeninges covering the base of the immunoreactivity to CAM5.2 low-molecular-weight keratin brain. Sections from the right basal ganglia and the internal cap- (prediluted, Becton-Dickinson, San Jose, CA, U.S.A.) and AFP sule showed irregular gliosis, edema, and perivascular infiltra- (1:50, Zymed, South San Francisco, CA, U.S.A.) antibodies and tion of lymphocytes and scattered atypical cells (Fig. 6). Multi- negative reaction to placental alkaline phosphatase (1:50, Dako, focal calcifications were also noted. Immunohistochemistry with Suprasellar Endodermal Sinus Tumor 437 A B C D Fig. 6. Microscopic examination of the right thalamus, basal ganglia, and internal capsule (A) shows irregular gliosis and edema with scat- tered atypical cells (arrows). Higher magnifications (B and C) show densely gliotic area with perivascular infiltration of atypical cells and floating atypical cells in loose edematous area (arrows). The immunohistochemical stain (D) shows glial fibrillary acidic protein-positive glial fibers with multifocal calcifications (arrows).
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