Journal of Neurology, Neurosurgery, and Psychiatry, 1975, 38, 493-499

Meningioma after contralateral hemispherectomy formalignant glioma: case report

P. J. E. WILSON AND D. J. B. ASHLEY

From the Departments of Surgical Neurology and Pathology,Morriston Hospital, Swansea

SYNOPSIS A patient is described who successfully underwent cerebral hemispherectomy formalignant glioma and whose death nine months later, wrongly ascribed to recurrent malignancy,was in fact due to a subsequently-developing benign meningioma in the remaining hemisphere. Thepossible advantages of a modification of standard hemispherectomy technique are also discussed.

Although the occasional concomitance of intra-cranial glioma with meningioma is well known,the following case is reported in detail becauseof unusual features in the diagnosis and manage-ment.

CASE REPORT

Mr D.B. (MH 31189/70) a married, right-handedshipping-clerk aged 33 years, was first referred to Dr

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B. M. Phillips, consultant neurologist, with a historyof epilepsy for the preceding 11 days.The patient had been perfectly well until the night

of 10 February 1970 when he suffered a nocturnaltonic seizure, witnessed by his wife, but for which hehimself was amnesic. A week later, while walking inthe street, he had sudden pain in the left foot spread-ing to involve the entire left side of the body; he fellto the ground, but did not convulse or lose conscious-ness, and was not incontinent. A similar attackoccurred on the day of referral, 21 February 1970.

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I n)FIG. 1 Rectilinzear brain scan on 22 April 1970, one month before right hemispherectomy. (a) Right lateralview, showing a diffuse area of increased isotope uptake in the parietal region. (b) Anteroposterior view, showingthat the increased isotope uptake is parasagittal and right-sided.

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Physical examination, skull radiographs, and EEGwere all entirely normal.During March 1970 he began to have increasingly

frequent and severe headaches, and experiencedseveral brief episodes of painful stiffness and par-aesthesiae in the left foot, sometimes also affectingthe left hand, and accompanied by an insidious lefthemiparesis.On readmission to hospital on 16 April 1970 (again

under the care of Dr B. M. Phillips), he was alert, ofnormal intelligence, with no papilloedema ormeningism, but had dense flaccid left hemiparesisand left-sided sensory impairment. An EEG nowrecorded persistent low-amplitude activity in thetheta and delta ranges from the right parietal leads.Right carotid angiography by Dr A. M. Jones on 20April 1970 showed deviation of the pericallosalarteries 6 mm to the left of the midline, but gave nodefinite localizing information. Brain scan on 22April 1970, using 99mTC, showed a poorly-definedzone of increased uptake in the right parietal para-sagittal region (Fig. 1). On 28 April 1970, lumbarpuncture yielded xanthochromic CSF containingprotein 65 mg/dl but only 4 red cells and 3 whitecells per mm3. He thereafter became insidiously moreinattentive and inert, and showed neck rigidity, buthis headaches and hemiparesis were unchanged. On9 May 1970, carotid angiography was repeated,showing that the pericallosal shift had increased to

14 mm, but again no abnormalities of localizingvalue were demonstrated.On 12 May 1970 he became frankly stuporose and

acute bilateral papilloedema was observed. Dexa-methasone 5 mg intramuscularly every six hoursrestored almost full alertness, and he was thenreferred to one of us (P.J.E.W.) on 13 May 1970. Thesame day, cerebral biopsy via a right parietal burr-hole yielded necrotic fragments of astrocytic glioma,grade II-III. On 14 May 1970, despite continued highdoses of corticosteroids, he relapsed into stupor.Accordingly, emergency right lateral craniotomywas performed. The brain was extremely tense, andthe pre-central parasagittal convolutions were partlyreplaced by soft pinkish tumour. All visible tumourtissue was excised. The resulting brain cavity exposedthe full depth of the falx and measured roughly7 x 4 x 4 cm. The surrounding brain was quite slack,but biopsy showed that tumour extended into itbeyond the anterior limit of the resection. Theexposed falx looked normal.The patient recovered promptly to a state of full

alertness. His hemiplegia showed no change. Becausethe criteria for radical surgery seemed to be satisfied(vide infra), the question ofcerebral hemispherectomywas discussed with both the patient and his relatives,who agreed to the procedure and all its implications.On 22 May 1970, the right cerebral hemisphere

was removed in one block. The line of section was

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FIG. 2 Rectilinear brain scan on 10 February 1971, nine months after right hemispherectomy. (a) Right lateralview, showing an area of abnormal isotope uptake of similar position, pattern, and density to that on the pre-operative scan. (b) Anteroposterior view, showing a general increase ofisotope uptake in the right hemicraniumand a dense circumscribed uptake in the left parasagittal region. This appearance is, in retrospect, consistentwith meningioma.

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extrathalamocaudate, mesial temporal structuresbeing left undisturbed. The ipsilateral choroid plexuswas coagulated. The septum pellucidum was notfenestrated. The calvarial dura mater was folded intothe right hemicranial cavity and sutured to the freeedge of the falx and tentorium so as to obliterate thepotential subdural space and 'screen' the thirdventricle. (These important departures from standardhemispherectomy technique will be discussed below).Detailed examination of the resected hemisphere wassomewhat hampered by the effects of operativevenous infarction but showed that the remainingtumour was everywhere contained within a generousmargin of normal brain.The patient's postoperative recovery was rapid and

smooth. He sat out of bed on the third day and wasable to walk with help on the tenth day. Slight

euphoria, fatuity, and social disinhibition were notedat times. Psychometric assessment on 29 June 1970gave him verbal-scale IQ 126, performance-scale IQ61 (WAIS). On 14 August 1970 he was allowed home.He was able to walk with the aid of a stick and a toe-raising device, but had virtually total spastic mono-plegia and sensory loss in the left arm. There was acomplete left homonymous hemianopia.

Intensive physical and occupational rehabilitationenabled him to return to part-time work in hisprevious job at the shipping agency, to which hewould travel by public transport. His fatuity re-solved, but his family commented that he was moreirritable, more self-centred, and less judicious thanbefore.He progressed well until January 1971, when he

noticed increasing difficulty in walking due to stiff-

FIG. 3 Ventriculography on I March 1971. (Topleft) Brow-up anteroposterior view, showing theleft frontal horn to be sllghtly enlarged and'ballooned' towards the empty right hemicranium.Between the sagittal burrhole and the lowest groupof metallic clips the sinuous line of the post-hemispherectomy duroplasty can be discerned. Aquantity of air remains in the right hemicraniumfrom a cavity air-sud erformed three days

-earlier (see tet). (Top right) Brow-up lateralview, showing slight depression of the body of theleft lateral ventricle. Air fills the pontine cistern.a;-;- _- An air-fluid meniscus is seen in the front of theright hemicranium. (Bottom left) Brow-downlateral view, showing normal size and configura-tion of the trigone and temporal horn of the left

lateral ventricle. The fourth ventricle has filled with air spontaneously, and is also normal in size, shape, andposition. The right hemicranial air-fluid meniscus has movedfreely to the back of the head.

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ness and dragging of the 'good' right leg. When re-admitted to hospital on 6 February 1971, he was alertand rational, not dysphasic, and had normal opticfundi. The postoperative left hemiplegia was un-changed, but there was global spastic monoparesis ofthe right leg, sensation in that limb being normal. AnEEG showed low-voltage activity over the righthemicranium, presumably transmitted from the left,and a non-focal increase of slower frequencies overthe left hemicranium. Repeat brain scan on 10February 1971 showed a dense but ill-defined area ofincreased uptake in the left central parasagittalregion, almost a mirror-image of the preoperativescan (Fig. 2). Left carotid angiography showedbowing of the pericallosal artery 21 mm to the rightof the midline, but no area of pathological circula-tion or other vascular displacements could be seen.On 26 February 1971 the right hemicranial cavitywas punctured and clear watery yellow fluid atnormal pressure was obtained (protein 7.2 g/dl,RBC nil, WBC 32 per mm3, mostly lymphocytes).Filtered air (30 ml) was injected into the cavity butnone of this could be induced to enter the remainingventricles. On 1 March 1971, via a left coronal burr-hole, pneumoventriculography was performed. Theleft lateral ventricle contained clear colourless CSF.There was a smooth depression and narrowing ofthe body of the ventricle, suggesting the presence of aparasagittal mass (Fig. 3). It was felt unlikely thatthe mass could be anything other than a gliomarecrudescence across the corpus callosum, and acannula biopsy on 8 March 1971 appeared to confirmthis view: material from the left hemisphere wasreported by one of us (D.J.B.A.) as follows: 'There

is extensive gliosis. There is one small area in whichthere is protoplasmic astrocytoma'.

Further surgery seemed unwarrantable in thecircumstances. Radiotherapy was considered butrejected. The patient showed a gradual decline intocoma and double hemiplegia, punctuated by severalgrand mal seizures. He died on 30 May 1971.Necropsy was performed by one of us (D.J.B.A.)

two days after death.The right cerebral hemisphere had been removed

surgically and the dura mater of the vault had beenfolded medially and sutured to the dura mater of thetentorium, and falx. Clear yellowish fluid occupiedthe space between the dura mater and the inner tableof the skull, and the transposed dura mater wasadherent to the remaining brain. The left cerebralventricle was compressed, and there were a fewminute granulations in its ependymal lining.A firm rounded tumour roughly 3 cm in diameter

was embedded in the medial surface of the leftparietal parasagittal cortex, and had its origin fromthe adjacent falx (Fig. 4). Histological examinationshowed this tumour to be a fibrous meningioma oftypical and benign appearance. Multiple sections ofthe brain substance showed gliosis round themeningioma. (It is probably from this zone that themisleading biopsy was taken). No gross or micro-scopic evidence of persistent glioma was found in theremainder of the brain. Sections of the wall of theventricle showed no ependymitis.

There was no noteworthy arachnoiditis and nomacroscopic staining of the surface of the brain byiron-containing pigments.

FIG. 4 Coronal section of theleft cerebral hemisphere, uppermid-brain and right basalganglia. The left hemisphere isswollen. An ovoid extrinsictumour with a whorled cut sur-face has invaginated the para-sagittal and parafalcine cortex.The left lateral ventricle is ofnormal cross-section,lbut the

W third ventricle is slightly7 dilated. A shrunken remnant of

the right hippocampus can beseen. (The specimen hasbecome somewhat distortedduring fixation.)

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DISCUSSION

This patient had two intracranial tumours, ofdifferent types, separated both spatially andanatomically. That on the right side, treated byradical surgery, was an intrinsic astrocytoma offibrillary and protoplasmic type, parts of itbeing Kernohan's grade II (relatively benign)whereas other areas were grade III (somewhatmore malignant). The tumour on the left sidewas a typical benign fibrous meningioma.Madonick et al. (1961) found that menin-

gioma and glioma was the commonest combina-tion of multiple diverse primary intracranialtumours.

Russell and Rubinstein (1971) record fiveinstances of this association which they hadthemselves observed. O'Connell (1961) reportedfour patients who had meningiomas in conjunc-tion with other central nervous system tumours:in one case, cranial and spinal meningiomas; inone case, meningioma and chromophobe pituit-ary adenoma; in one case, meningioma andacoustic neuroma; and in one case, meningiomaand astrocytic glioma. A recent monographbased on McKissock's personal series of 916parasagittal and falx meningiomas includes one

instance (case 130) of astrocytic glioma of thecerebellum in association with a parietal para-sagittal meningioma (Gautier-Smith, 1970). Ifwe exclude the various phakomatoses-notably,von Recklinghausen's disease-such combina-tions of different types of primary intracranialtumour are probably more common than isactually recorded in the literature; one of us

(D.J.B.A.), with a much smaller personalexperience than Russell and Rubinstein, hasencountered four cases, three of concomitantmeningioma and glioma, and one of concomitantmeningioma and acoustic neuroma.

In most instances, the two tumours have beenspatially or anatomically separated. It is thusdifficult to conceive of a common local onco-

genic stimulus. One could postulate loosely thatthe presence of one intracranial tumour may insome way alter the environment of other partsof the brain and thereby lead possibly to a situa-tion in which cells are more easily stimulatedinto new growth. Our patient did not receiveradiotherapy, but had he done so it would havebeen necessary to consider whether ionizing

radiations may have induced a neoplasticpropensity in mesodermal tissues. Norwood etal. (1974) describe two examples of benignconvexity meningioma presenting 25 and 15!years after irradiation for optic nerve glioma andhind-brain glioma respectively. Munk and hiscolleagues (1969) reported one presumed andfour proved examples of meningioma occurringin patients whose skulls had been irradiated forringworm. McKissock's case, already men-tioned, had had radiotherapy for his cerebellarastrocytoma 15 years before the onset of symp-toms attributable to his meningioma (Gautier-Smith, 1970). Examples of glioma after radio-therapy seem to be very rare. Butler and Netsky(1973) averred that none had yet been docui-mented in man. However, one such case isdescribed by Jones (1960), without comment asto any possible link between irradiation andoncogenesis. This was a patient who had alesion-dose of 4 000 r to an inoperable angio-blastic meningioma of the left frontal region,and died 10 years later from an astrocytoma ofthe opposite hemisphere, the original menin-gioma having regressed and become psammoma-tous.The ideal treatment for malignant cerebral

glioma is total surgical extirpation with a widemargin of uninvolved but dispensable brain-tissue. In practice, it is rare for this ideal to beapproached except at the risk of crippling neuro-logical sequelae. Even more rarely can theneurosurgeon feel justified in offering his patienta reasonable guarantee of cure at the bitter priceof deliberately incurring an irrevocable neuro-logical deficit.

However, since Dandy's first reports nearlyhalf a century ago, cerebral hemispherectomy(the ultimate weapon of radical brain surgery),has from time to time been employed, with aproportion of good long-term results (Dandy,1928; Bell and Karnosh, 1949; Gardner, 1954;Gardner et al., 1955).The indications for this severe procedure in

the treatment of glioma are not absolute, butmany surgeons would accept the following: thetumour should be proven low-grade gliomasuperficially-placed in the Rolandic area of thenon-dominant hemisphere in a young patient ofgood intelligence and pre-morbid personality,who has a stable domestic background, an

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adaptable occupation, can rely upon fullresources for physical and mental rehabilita-tion, and who already has an established andirreversible hemiplegia.The initial result in our patient was encourag-

ing. Despite a severe fall-off in performancescale sub-tests he was able to return to paidclerical work, and achieved a near-normalmeasure of personal independence. The lesstangible psychological legacies of hemispherec-tomy, stressed by Gardner et al. (1955) werepresent, but not to a distressing or disablingdegree.When, some eight months after hemispherec-

tomy and despite the histological evidence ofcomplete removal of tumour, the patientdeveloped progressive symptoms and signsimplicating the remaining hemisphere, it seemedthat the most likely explanation was a recrudes-cence of the original glioma via the corpuscallosum into the contralateral Rolandic area.Investigations were all in ostensible support ofthis assumption, and the possibility of an inde-pendent benign lesion was not seriously con-sidered, especially after biopsy was construed aspositive for glioma. In retrospect, the biopsycannula had merely skirted the meningioma butnot sampled it.The postmortem appearances make it clear

that, had the meningioma been successfullybiopsied, its total removal would have presentedless technical difficulty than usual, by a trans-falcine approach through the empty right hemi-cranium. The clinical outcome should then havebeen a happier one. The commonest cause ofmorbidity and later mortality after hemispherec-tomy (for whatever indication) is chronic sub-dural haemorrhage into the hemicranial cavity,resulting in granular ependymitis, obstructive orcommunicating hydrocephalus, and-in a fewcases-subpial haemosiderosis of the brain andspinal cord (Griffith, 1967; Falconer and Wilson,1969; Wilson, 1970). Many early descriptions ofthis sequence of events were published in greatdetail before their pragmatic implications wererealized. Not only is this syndrome of delayedintracranial haemorrhage treatable, it should belargely or entirely preventable. In the case nowreported, a deliberate modification of thestandard hemispherectomy technique was em-ployed. It has been customary, after removal of

the hemisphere, to fenestrate the septum pelluci-dum and to coagulate and excise the remainingchoroid plexus, with the idea of preventingbrain-stem shift in the immediate post-surgicalphase (Cabieses et al., 1957) and of minimizingthe development of hydrocephalus in the laterconvalescent period (McKissock, 1953). Thisstep in the technique may do more harm thangood, because it enables the large, potentiallyblood-filled subdural cavity to drain unimpededinto the ventricular and subarachnoid CSF path-ways. The basic dangers of such a 'sump' effecthave long been recognized in other surgical con-texts (Finlayson and Penfield, 1941), and theterm 'acute postoperative aseptic leptomenin-gitis' was coined to describe the stormy coursethat often follows such a situation. The modifica-tion used in the present case was to screen theventricular system from the operative cavity byfolding the vault dura mater down into it andsuturing it firmly to the falx, tentorium, andbasal dura mater. The patient's surgical con-valescence was very smooth, without pyrexia,meningism, or headache. Although (extradural)cavity puncture eight months after operationyielded yellow and highly proteinaceous fluid,the ventricular CSF at that time was in strikingcontrast, being clear and colourless, with a pro-tein level of only 80 mg/dl, WBC 1 per mm3,RBC 1 600 per mm3 (the latter probably oftraumatic origin). No neuropathological evi-dence of significant ependymitis or arachnoiditiswas found when the brain was examined afterfixation.We feel that our failure to suspect a benign

nature in the second tumour, compounded bymisinterpretation of the biopsy from brain tissueclose to the meningioma, determined the fataloutcome. It is worth keeping in mind the possi-bility of a second and different tumour when apatient with a known intracranial neoplasmdevelops symptoms indicative of recurrence,especially if these symptoms suggest an unusualmode of recrudescence or spread.

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Butler, A. B., and Netsky, M. G. (1973). Classification andbiology of brain tumors. In Neurological Surgery, vol. 3,

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