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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