recurrent intracranial meningioma with malignant …...malignant meningioma involving bilateral...
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Journal of tbe Korean Radiological Society 1995: 33( 1) ‘ 55 - 58
Recurrent Intracranial Meningioma with Malignant Change and Extracranial Bone Metastasis :
A Case Report1
Nak Kwan Sung, M .D ., Yeong Hwan Lee, M.D.
In general , meningiomas are slowly growing benign neoplasms originating from specialized meningothelial cells in arachnoid granulation, but have a tendency to be locally invasive and recurrent. Meningiomas very rarely metastasize outside the nervous system, occurring in less than 0.1 %.
We report the CT and MR findings of a case of a solitary benign syncytial meningioma showing recurrent multiple tumors and malignant progression with eventual bone metastasis to rib after six su rgical extirpations duri ng six years.
Index Words: Meninges, neoplasms Meninges, MR Bone neoplasms, secondary
Less than 9 % 01 intracranial meningiomas represent multiple lesions and only 1 -2 % are anaplastic or malignant(1 , 2). It has been estimated that less than 0.1 % 01 meningiomas metastasize, and both the benign and malignant meningiomas can metastasize to distant sites(3,4)
This is a case 01 m비tiple and malignant intracranial meningiomas metastasizing to rib , which progressed Irom a solitary benign syncytial one after six times 01 surgical extirpation during six years
CASE REPORT
A 48 -year - old man presented with several - month history 01 progressive right hemiparesis and dull headache. CT revealed a large lobular mass 01 9 cm in maximal diameter in the left high parasagittal region abutting the lalx. The mass was hyperdense and diffusely enhanced with a small area 01 central low density(Fig. 1 a). He underwent his lirst operation lor total removal 01 the mass and it was lound to be a well encapsulated hard mass tightly adherent to the left side ollalx and superior sagittal sinus. No evidence 01 underlying brain parenchymal invasion was noted. Pathologic diagnosis was benign syncytial meningioma(Fig. 1 b).
'Department 01 Di agnoslic Rad iology, School 01 Medicine , Taegu Calhol ic Uni. versity Received M ay 11,1995; Accepted June 16,1995 Address reprinl requests 10: Nak Kwan Sung, M.D., Department 01 Diagnostic Radiology, School olMedicine, Taegu Catholic University ~ 3056.6, Taemyung.4 dong , Nam.gu, Taegu , 705-034 Korea
Te l. 82.53.650. 41 03 Fax. 82- 53. 621- 41 06
One year and 8 months later , the second operation was perlormed to remove a recurrent tumor in the same site. Pathologic diagnosis was atypical meningi oma. Two more operations were done during the next 3 years to remove the recurrent histologically proven atypical meningiomas.
One year after the 4th operation , the patient was , readmitted because 01 aggravating right hemiparesis. FOllow - up CT demonstrated a recurrent cystic mass with two large enhancing nodules , but there was no enhancement in the cyst wal l. On MR(Fig. 2a -c) , the cyst Iluid was hyperintense on both T1 -weighted (T1 WI) and T2 -weighted images (T2W/) suggesting Iysed chronic hematoma, and the wall was isointense on T1 WI and hypointense on T2WI suggesting to be laden with hemosiderin. The solid nodules showed isosignal inten-sity on T1WI and slightly high signal intensity on T2WI , and rather heterogeneous contrast enhancement was seen. There was another small Ilat extraaxial solid mass at right lower Irontotemporal convexity having signal characteristics similar to the left parasagittal nodules. The lifth operation was performed to remove the recurrent left parasagittal mass. The cyst Ilu id was shown to be liquelied darkish hematoma surrounded by a yellowish capsule and underlying brain paren-chyma was invaded by the tumor. A histologic examination demonstrated malignant meningioma with aty-pical meningothelial cells , cellular pleomorphism , Ire-quent mitoses, local necrosis and invasion 01 brain , and the capsule contained a lot 01 hemosiderin (Fig. 2d).
One year later , he was readmitted lor tonic -clonic
야 μ
Journal of the Korean Radiological Society 1995; 33( 1) : 55-58
a b
seizure and paraparesis. A follow - up CT demonstrated a recurrent tumor invading and penetrating the falx He underwent his 6th operation to remove the recurrent malignant meningioma involving bilateral parasagittal reglons.
One year after the last operation , a follow -up CT revealed local recurrence in the left high parietal region , more enlargement of the right frontotemporal convexity mass with calcifications , and two new growths were seen abutting the superior border of the right tentorium and the le付 border of anterior falx(Fig. 3). Chest CT revealed the left 4th rib destruction with a heterogeneously enhancing bulky soft tissue mass and a histolog ic examination demonstrated metastatic malignant meningioma(Fig. 4).
Despite 6 surgical extirpations of the recurrent meningiomas progressing from a typical benign one to an atypical and malignant one , the patient died about 8 years after the initial presentation
DISCUSSION
Although meningiomas usually are considered to be benign and cause compression of adjacent neural tissue , there are cases in which they show local invasion to dura , dural sinus , bone , muscle and brain substance. And local tumor recurrence has been reported to occur in 9% to 32% of cases depending on the follow - up periods(5 , 6). Reports suggest that recurrence depends on the incompleteness of tumor excision. It is well known that such local invasion and recurrence are not necessarily indicative of malignancy, although malignant meningiomas account for much higher incidence of local invasion and recurrence
Of the meningiomas, 1 - 2% are malignant(2). His-
ν:~ .~ ‘ Fig. 1. Benign syncytial meningioma at in 양 itial presentation.
훌혹 a. PO떠s앉t-contr때tr때ra없s앙tαtCTσTs앙해ho애ws a히|녀때a하rge’ d미이ifflωu뼈J ’ 겁 enhanci매 lobular mass with a central low ~ density in left high parasagittal region abut겨 tingthelalx
b. M icroscopic leatu res show typical beni gn syncytial meningioma with sheets 01 men ingothel ial cells , round to ovoid nuclei , delν cate chromatins , abundant cytoplasm and exceptional mitosis(H&E , X400)
tologic criteria for malignant meningiomas remain at least partly uncertain. Greater than 10 mitoses per 10 high - power fields and atypical features are the most reliable indicator of malignant behavior , but that measure is also impertect. Rapid rate of recurrence , aggressive local invasiveness and the presence of remote metastases are also suggested for determ ining the criteria of malignancy. But as described previously, the former two have a variable and significant margin of error in predicting malignant biologic behavior. Di Chiro et al. (7) demonstrated that positron emission tomography with fluorine-18 -2 - fluorodeoxyglucose is reliable for predicting the biologic behavior and recurrence of meningiomas by assessing tumoral glucose utilization
As in our case , parasagittal meningiomas are known to have greater probability of recurrence and malignant change than those in other locations. Reported CT findings favoring malignancy of meningioma include extensive bone destruction , irregular indistinct tumor marg ins , deeply penetrating fronds , extensive necrosis , absent or minimal calcification and mushrooming pannus of tumor extending from the globoid part of the tumor. But there are few reports about MR findings of malignant meningiomas(8). MRI of our case showed extratumoral cystic pattern of malignant meningioma after 4 surgeries , and the cyst resulted from Iysed chron ic hematoma. Cystic meningiomas have two different morphologies: intratumoral and extratumoral cysts. Intratumoral cysts representing tumor necrosis or degeneration show rim enhancement. On the other hand , extratumoral cysts representing reactive arachnoid cyst, direct secretion of fluid by tumor cells , absorption of hemorrhage as in our case , or loculated cerebrospinal fluid show no rim enhancement(9)
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Nak Kwan Sung, et al: Recurrent Intracranial Meningioma with Malignant Change and Extracranial Bone Metastasis
a b
c d
a b
런 기
Fig. 2. Recurrent cystic meningioma with
malignant progression after 4 times of sur-
gical extirpation.
a, b. The cystic component is hyperintense
on both T1WI(a , TR/TE=650/25) and T2WI(b,
TR /TE=2300/520) suggesting chronic Iysed
hematoma and the wall(arrows) is hypoin
tense on T2WI(b) suggesting hemosiderin
deposit
c. Post-contrast T1 WI shows diffuse en-
hancement of the masses
d. Microscopic features show findings of
malignant meningioma with sheets of aty
pical meningothelial cells , cellular pleom
orphism , hyperchromatism and frequent
mitoses(H&E , X400)‘
Fig. 3 . Recurrent and multiple menin
giomas after 6 times of surgical extirpation
a. Post-contrast CT shows a large recurrent
mass in left high parietal region and another
small mass is seen abutting left anterior
falx
b. Lower level scan shows two more tumors
at right frontotemporal convexity and upper
border of right tentorium. Note calcifications
in rightfrontotemporallesion(arrows)
Journal of the Korean Radiological Society 1995; 33( 1) : 55 - 58
Fig. 4. Metastatic meningioma Post-contrast CT 01 chest shows destruction 01 left posterior 4th rib with a heterogeneously enhancing bulky soft tissue mass
Multiple meningiomas occur in less than 9% of im
aged cases and are known to have an identical his
tologic type in each case(1). Although multiple meni
ngiomas can be associated with neurofibromatosis
type 2, the majority of patients have no relation with it
Our patient also showed no other evidences of neu
rofibromatosis. The loss of genetic material in chromo
some 22 has been proposed as a cause of multiple
menmglomas.
Distant metastasis of intracranial meningiomas is
extremely rare and is said to arise in less than one in
1000(3, 4 , 1 이 The malignancy of a tumor is usually
defined by its ultimate biologic behavior, that is , by its
propensity to metastasize. But not only the malignant
but also the atypical and benign meningiomas have
been reported to metastasize via blood stream and cer
ebrospinal fluid pathway(3 , 4 , 8 , 11). Those benign
metastasizing mening iomas could be due to highly pro
liferative activity of the neoplastic cells. Most common
sites of metastases are lung , liver and Iymph nodes.
The hematogenous spread via the caval circulation fol-
lowing invasion of dural sinuses probably explains the
high incidence of pulmonary metastasis(11). Bony me
tastasis as in our case has been reported to constitute
about 20% of all metastases(3 , 4). There has been co
ntroversy about the relationship between metastatic
disease and surgery. Although about 75% of reported
cases metastasized after variable number of surgical
extirpations , no particular relationship is known to be
present between them(12)
REFERENCES
1. Lusin JO, Nakagawa H. Multiple meningiomas evaluated by computed tomography. Neurosurgery 1981 ; 9: 137-141
2. Maier H, ffer D, Hiffmair A, et al. Classic, atypical and anaplastic meningioma: three histopathologic subtypes 01 clinical relevance. J Neurosurg 1992; 77 : 616-623
3. Shuangshoti S, Hongsaprabhas C, Netsky MG. Metastasizing meningioma. Cancer 1970 ; 26: 832-841
4. Wasserkrug R, Peyser E, Lichtig C. Extracranial bone metastasis from intracranial meningiomas. Surg Neuro/1979; 12: 480-484
5. Mirimanoff RO , Dosoretz DE, Li nggood RM , et al. Meningioma analysis 01 recurrence and progression lollowing neurosurgical resection. J Neurosurg 1985 ; 62 ‘ 18-24
6. Adegbite AB , Khan MI , Paine KWE, Tan LK. The recurrence 01 intracranial meningiomas after surgical treatmen t. J Neurosurg
1983 ; 58 : 51-56 7. Di Chiro G, Hatazawa J, Katz DA, Rizzoli HV, De Michele DJ. Glu
cose utilization by intracranial meningiomas as an index 01 recurrence: a PETstudy. Radiology 1987; 164: 521-526
8. Greenberg SB , Schneck MJ , Faeber EN , Kanek PM. Malignant meningioma in a child: CT and MRI lindings. AJR 1993 ; 160 1111-1112
9. Worthingtom C, Caron J, Melanson D, Leblanc R. Meningioma cysts. Neurology 1985; 35 ‘ 1720-1724
10. Kang HJ, Eun CK , Han CY. The malignant meningioma with extracranial metastasis. KJR 1979 ; 15 ; 311-317
11. Som PM , Sacher M, Strenger SW, Biller HF, Malis LI “Benlgn metastasizing meningiomas. AJNR 1987; 8; 127-130
12. Russel DS, Rubinstein LJ. Pathology of tumors of the nervous
system. 5th ed. Baltimore; Williams and Wilkins 1989 ; 504-529
대 한 밤 사 선 의 학 회 지 1995 ; 33( 1) ; 55-58
악성변화와 두개외 골전이를 일으킨 재발성 뇌수막종 :1예 보고1
1 대구효성가톨릭대학교의과대학진단방사선과학교실
성 낙 관·이 영 환
일반적으로 뇌수막종은 거미막과립의 특수 수막세포에서 기인하여 서서히 자라는 앙성종앙이지만 국소적으로 침습적이
고 재발하는 겸향이 있다. 노|수막종이 신경계 밖으로 전이를 일으키는 경우는 0.1 %이하로 매우 드물다.
여섯 번의 외과적 제거술 후 고립성의 양성합포체성 뇌수막종이 다발성의 악성으로 진행하여 늑골로 전이를 일으킨 예의
CT와 MR소견을 보고한다.
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