recurrent subarachnoid haemorrhage paraventricular lesions with normal angiography ·...

J. Neurol. Neurosurg. Psychiat., 1964, 27, 435

Recurrent subarachnoid haemorrhage fromparaventricular lesions with normal

angiographyR. M. KALBAG

From the Midland Centre.for Neurosurgery and Neurology, Smethwick

Vascular anomalies, either aneurysmal or arterio-venous, figure predominantly as the causative lesionsin spontaneous subarachnoid haemorrhage. Suchhaemorrhage as the presenting feature of intra-cranial tumour, although much rarer, is sufficientlywell recognized not to cause diagnostic difficulties; _ ,Jablost invariably some evidence of a space-occupy-ing lesion, if not of a pathological circulation, isseen on subsequent angiography, especialy in casesof supratentorial neoplasm. Ernsting (1955) reporteda patient with recurrent subarachnoid bleeding, inwhom, after carotid angiograms a year previouslyhad been regarded as normal, a choroid plexus papil- aloma was demonstrated when the investigation wasrepeated. The lesion was confirmed by air studiesand successfully removed. Ernsting pointed out thepossibility of a small papilloma bleeding into theventricle and yet not being apparent on arterio-grams.The purpose of this paper is to present three cases

with recurrent haemorrhage from paraventricular FIG. 1 Case: Normaleantero-posteriorarterlesions not revealed on angiography. compression ofthe kft carotid artery in the ned

CASE REPORTS

CASE 1 T.B., a man aged 37 years, had had a subarach-noid haemorrhage in 1949, after which he was un-conscious for three weeks. He developed a left hemiplegiawhich improved slowly. Eventually, despite slightm-emory impairment, he was able to return to his formeroccupation as an instrument maker. In June 1958 he had asecond haemorrhage, without loss of consciousness, ac-companied by an increase in the left hemiparesis, whichwas maximal in the arm. Lumbar puncture confirmed thediagnosis.He was admitted to the Midland Centre for Neuro-

surgery on 15 February 1960, when he still had a minimalspastic left hemiparesis and a complete left homonymoushemianopia. The fundi were normal.

Radiographs of the skull were normal apart from anosteoma in the right frontal sinus. Bilateral carotidangiograms (Figs. 1 and 2) were also normal. As the right FIG. 2. Case 1: Normal lateral arterial phaposterior cerebral vessels filled on carotid injection, carotid injection.

435

iogram withkT.

7se on right

Protected by copyright.

on July 7, 2020 by guest.http://jnnp.bm

j.com/

J Neurol N

eurosurg Psychiatry: first published as 10.1136/jnnp.27.5.435 on 1 O

ctober 1964. Dow

nloaded from

http://jnnp.bmj.com/

R. M. Kalbag

FIG. 3. Case 1: Air ventriculogram (reverse Towne'sview)showing irregularfilling defect in the wall ofthe right lateralventricle posteriorly.

FIG. 4. Case 1: Air ventriculogram (lateral prone film)showing irregularity in the wall ofthe right lateral ventricle.

vertebral arteriography was considered unnecessary inthe presence of the precise clinical localization of thelesion.The cerebrospinal fluid was normal.In view of the negative arteriography, a good prognosis

was given with respect to recurrent bleeding, and twomonths later he returned to work.On 13 July he was admitted to a general hospital after

a fresh subarachnoid haemorrhage, when the neurologicalfindings were again a complete left homonymoushemianopia and a left hemiplegia. There was clinicalevidence of two further bleeds while he was still inhospital.

He was transferred to the Midland Centre for Neuro-surgery on 8 September. Bilateral carotid angiograms wererepeated, and on this occasion the only abnormal findingwas a slight deep venous shift to the left. A right vertebralarteriogram was normal. An air ventriculogram (Figs.3 and 4), however, showed widespread irregularities ofcontour of the ventricular walls, maximal on the right,posteriorly. The ventricular fluid was xanthochromic.On 1 November an exploratory right postero-lateral

craniotomy was performed by Mr. J. G. Hamilton. Hefound yellowish staining around the posterior end of theSylvian fissure. A core of brain was removed from thetemporo-parietal region to expose soft gelatinous tissueof moderate vascularity at a depth of 1.5 cm. continuingdown to the ventricle. On looking into the ventricle itself,similar tissue was seen lining it in all directions. In viewof the vascularity, most of this was left undisturbed,the core of tissue obtained being sent to the laboratory.The pathologist reported that the lesion was undoubtedlyan oligodendroglioma, though as tissue approached theventricle the cells became increasingly vacuolated anddegenerate.

After the operation, the patient remained unchangedneurologically, and was transferred back to the originalhospital, where he progressively deteriorated and died afew months later.

CASE 2 C.R., a girl aged 7 years, at the beginning of May1959 developed a severe headache, vomiting, and neckstiffness, without loss of consciousness. It was not certainwhether this headache was of sudden onset. Five dayslater she was admitted to a general hospital, wherelumbar puncture yielded xanthochromic cerebrospinalfluid. Her symptoms eased spontaneously within a fewdays.On transfer to the Midland Centre for Neurosurgery

on 9 June 1959, apart from a possible right lower facialweakness, there were no abnormal physical findings.

Radiographs of the skull were normal, and bilateralcarotid angiography showed no convincing vascularanomaly or displacement (Figs. 5 and 6).

After discharge, she had further episodes of recurrentheadache, vomiting, and neck stiffness; the cerebro-spinal fluid examined some days after one of these attackswas again yellow.

She was readmitted on 26 March 1960 four weeks afterthe last attack of headache. This time there was someblurring of disc margins with a subhyaloid haemorrhageon the right. On 2 April she complained of a suddensevere frontal headache, followed by vomiting, photo-phobia, and drowsiness, with marked neck retraction anda positive Kernig's sign, but no focal neurologicalabnormality.

Ventriculography (Figs. 7 and 8) showed a mainlyright frontal space-occupying lesion, indenting theanterior horn and the anterior end of the third ventricle.On 6 April a right frontal craniotomy was performed byMr. J. M. Small, and a bluish intrinsic neoplasm withyellowish staining and a somewhat nodular capsule wastotally excised.The greater part of the specimen consisted of very large

blood spaces containing partly organized thrombus.

436



j.com/

J Neurol N


ctober 1964. Dow

nloaded from


Recurrent subarachnoid haemorrhage from paraventricular lesions with normal angiography

n . St'L' t \, '~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~~gielAS9iSEW02fiLljilE'ySiiGis -{'SESiiti,/Z,gi,z,,U,,,,f'

437

FIG. 5

FIG. 8

FIGS. 5 and 6. Case 2. Antero-posterior and lateralarterial phases of right carotid angiogram.

FIGS. 7 and 8. Case 2: Air ventriculogram to demonstratea mainly right frontal space-occupying lesion indenting theanterior horn andfront end ofthe third ventricle.

FIG. 9. Case 3: Prone lateral air ventriculogram out-lining an air-filled cavity above the posterior end of thethird ventricle.

FIG. 9



j.com/

J Neurol N


ctober 1964. Dow

nloaded from


R. M. Kalbag

There was also evidence of old haemorrhage in thesurrounding tissue and of a glial reaction to this. Thepicture was that of an angiomatous malformation ratherthan a neoplasm.She was discharged home on 29 April, having re-

covered completely. She has remained well since.

CASE 3 L.A., a boy aged 13 years, on 28 February 1960,while out cycling with his father, developed sudden severeheadache, vomited, and shortly afterwards lost con-sciousness. He was admitted to a general hospital deeplyunconscious. Lumbar puncture yielded uniformly deeplyblood-stained cerebrospinal fluid, but neck stiffness and apositive Kernig's sign were not noted till the followingmorning. He regained consciousness some 12 hours afteradmission, but remained drowsy, restless, and irritablewith an increasing neck stiffness, and a pulse of 60/min.On 4 March he was transferred to the Midland Centre

for Neurosurgery, where, apart from meningism, noabnormal neurological signs were found.

Bilateral carotid angiography showed no evidence ofany supratentorial space-occupying lesion or vascularanomaly. The configuration of the anterior cerebralvessels, however, suggested enlarged lateral ventricles.

Parietal burr holes were made. Ventricular pressurewas high, and the cerebrospinal fluid was stained darkbrown with old blood, especially on the left side. Withventricular drainage, his headaches and level of con-sciousness gradually improved.On 9 March an air ventriculogram was carried out:

the ventricular size and pressure were now normal, andfilms showed a small space-occupying lesion with an air-filled cavity above the posterior end of the third ventricle,causing some distortion in the region of the trigone ofboth lateral ventricles, perhaps more so on the left (Fig.9).

Because of the site of the lesion and the spontaneousimprovement, no surgery was undertaken.The patient remained fit and symptom free until

18 March 1962, when he felt generally unwell with anattack of 'shivering' lasting about 15 minutes, followedby severe headache and nausea without loss of conscious-ness. He had several such attacks in the next three days,in the course of which he also developed neck stiffness.He was admitted to the Midland Centre for Neuro-

surgery on 27 June fully conscious with slight photo-phobia and neck stiffness and coarse horizontal nystag-mus on lateral gaze, slight right lower facial weakness,bilateral extensor plantars.On the evening of admission, he developed another

severe headache with marked drowsiness, opisthotonos,and shaking of all limbs, but improved considerably afterthe right lateral ventricle had been tapped, releasingheavily blood-stained fluid under high pressure.

Vertebral angiography was normal. Air ventriculo-graphy again showed the small air-filled cavity in the roofof the posterior end of the third ventricle, but there wasmore marked generalized distension of the lateral ven-tricles, and no air passed into the subarachnoid spaceabove the tentorium. Screening with Myodil confirmedthat the cavity in the 'tumour' communicated with thethird ventricle.

The lesion was presumed to be of a congenital nature,probably a vascular hamartoma, not readily accessibleto surgery. Following spontaneous full recovery he wasdischarged home on 27 July.

DISCUSSION

In all published series on subarachnoid haemorrhage,there is a proportion of cases in which no source ofbleeding has been found. Bjorkesten and Troupp(1957), comparing the ultimate fate of 40 cases withproven aneurysms treated conservatively, as againstthat of 61 in which no abnormality was demonstratedon bilateral carotid arteriography, concluded thatthe outlook as to a fatal recurrence was 10 timesbetter in the latter group than in patients with knownaneurysms not subjected to surgery. The proportionof negative investigations is, however, reduced whenvertebral angiography has been carried out in addi-tion. Spatz and Bull (1957) reported 60 cases, in16 of which anomalies were demonstrated on ver-tebral injection after having had normal bilateralcarotid arteriograms. It is now generally acceptedthat where total cerebral angiography has beennormal, the likelihood of a fatal recurrence iscomparatively remote.The prognosis on the other hand is undoubtedly

worse in the case of haemorrhage from tumours.Glass and Abbott (1955) reviewed the literature onsubarachnoid haemorrhage 'consequent to intra-cranial tumour'. They analysed 41 cases with recordscomplete enough to be used for the study. Fatalbleeding in the initial attack was found in threeinstances, while in the majority of cases multiplebleeds with a fatal outcome were observed. Of theirseven cases, only two had had arteriography, whichrevealed the source of the bleed; while five had pre-ictal symptoms pointing to a tumour as the probableorigin. Neoplasms lying in or alongside the ventriclesare particularly liable to present as subarachnpidhaemorrhage without, at the same time, bleedinginto the brain substance to form localized haema-toma, and consequent vessel displacements visiblearteriographically. Arterial displacement in the'absence of intracerebral clot may also occur,especially in the vicinity of the trigone when thetumour is large enough to obstruct the temporal!horn which later distends. In one of the two cases ofintraventricular meningioma described by Askenasyand Behmoaram (1960), while there was no vesseshift in the antero-posterior views, the middle cere-bral group was raised in the lateral films. Mille(1961) reported a case with recurrent episodes oheadache, vomiting, photophobia, and neck stiffnessin which subarachnoid haemorrhage was confirmedon lumbar puncture and the initial angiograms were

438



j.com/

J Neurol N


ctober 1964. Dow

nloaded from


Recurrent subarachnoid haemorrhage from paraventricular lesions with normal angiography

normal. Some four months later following anotherattack and again normal angiograms, presumably be-cause of a defect of conjugate upward movement ofthe eyes, an air ventriculogram was carried out toshow a space-occupying lesion in the region of thepineal, which eventually proved to be a cavernous

haemangioma involving the cistern of the greatcerebral vein.

Fine, Paterson, and Gaylor (1960) described re-

current subarachnoid haemorrhage in the presence

of an angioma associated with an intraventricularoligodendroglioma. They could not find any otherinstance of an oligodendroglioma bleeding spon-

taneously. In view ofthe coexistent vascular anomaly,however, it was difficult to decide with any certaintywhich of the two lesions was responsible for thehaemorrhage.While accepting the rare possibility of an oc-

casional tumour being missed in cases of subarach-noid haemorrhage with normal angiograms, routineventriculography would be impracticable andsuperfluous in the relatively large numbers of suchcases any neurosurgical unit may have to cope with.Some attempt at establishing criteria for carryingout this procedure is therefore desirable. In thiscontext, the term 'angiography' is deliberately pre-

ferred to 'arteriography' to emphasize an attentionto all phases of the cerebral circulation. At thishospital, the investigation routinely comprises atleast four views in the lateral series and three in theantero-posterior.The presence ofpapilloedema or focal neurological

deficit following an intracranial haemorrhage are

common enough in the aneurysm group as not tojustify in themselves routine ventriculography insubarachnoid haemorrhage. On the other hand,from the prognostic point alone, if for no other

reason, where, in cases of recurrent subarachnoidhaemorrhage, angiography has failed to reveal thesource of the bleed, air studies seem to be justifiedin the hope of excluding the rare, sometimes oper-able, paraventricular neoplasm.

SUMMARY

Three cases of recurr_nt subarachnoid haemor-rhage are reported in which angiography showed noabnormality but where lesions in or alongside theventricles were revealed on ventriculography. It issuggested that perhaps the investigation in all in-stances of recurrent haemorrhage may bring to lightmore such tumours than has hitherto been the case.

I wish to thank Mr. J. M. Small for his interest andadvice, and the consultants of the Midland Centre forNeurosurgery for permission to report on their cases.

REFERENCES

Askenasy, H. M., and Behmoaram, A. D. (1960). Subarachnoidhemorrhage in meningiomas of the lateral ventricle. Neurology(Minneap.), 10, 484-489.

Bjorkesten, G. af., and Troupp, H. (1957). Prognosis of subarachnoidhaemorrhage. J. Neurosurg., 14, 434-441.

Ernsting, J. (1955). Choroid plexus papilloma causing spontaneoussubarachnoid haemorrhage. J. Neurol. Neurosurg. Psychiat., 18,134-136.

Fine, R. D., Paterson, A., and Gaylor, J. B. (1960). Recurrent attacksofsubarachnoid haemorrhage in presence ofa cerebral angiomaand an intraventricular oligodendroglioma. Scot. med. J., 5,342-346.

Glass, B., and Abbott, K. H. (1955). Subarachnoid haemorrhageconsequent to intracranial tumours-Review of literature andreport of 7 cases. Arch. Neurol. Psychiat., 73, 369-379.

Miller, R. H. (1961). Spontaneous subarachnoid hemorrhage: apresenting symptom of a tumor of the third ventricle. Surg.Clin. N. Amer., 41, 1043-1048.

Spatz, E. L. and Bull, J. W. D. (1957). Vertebral arteriography in thestudy of subarachnoid haemorrhage. J. Neurosurg., 14,543-547.

439



j.com/

J Neurol N


ctober 1964. Dow

nloaded from


recurrent subarachnoid haemorrhage paraventricular lesions with normal angiography ·...

Documents