Journal of Neurology, Neurosurgery, and Psychiatry, 1979, 42, 1050-1053

Pineal apoplexyK. HIGASHI, S. KATAYAMA, AND T. ORITAFrom the Department of Neurosurgery, Yamaguchi University School of Medicine, Ube, Japan

S U M MARY A case of haematoma in the pineal region is reported in a patient whose initialsymptom was subarachnoid haemorrhage. The encapsulated haematoma and surrounding bloodclots were removed surgically. Haemorrhage into a pineal cyst may have been the cause of thisparticular intracranial mass.

Although pineal tumours are reported to be notuncommon in Japan (Katsura et al., 1959; Arakiet al., 1969), haemorrhage into the pineal tumour,pituitary apoplexy, has been a rare occurrence.Apuzzo et al. (1976) recently reported a case ofpineal apoplexy caused by haemorrhage into apineal cyst in a patient on anticoagulant therapy.We report a case of haematoma in the pinealregion developing without any anticoagulanttherapy.

Case report

A 51 year old previously healthy woman experi-enced the sudden onset of severe headache and abrief period of unconsciousness. On admission tothe local community hospital, spinal tap revealedgrossly bloody spinal fluid with an opening pressureof 240 mmH2O. The headache, nausea, andvomiting continued for three weeks and thengradually subsided.On admission on 27 June 1978, the patient was

alert. Her blood pressure was 140/80 mmHg.There was marked bilateral papilloedema withretinal haemorrhage. Pupillary light reflexeswere sluggish bilaterally, without impairment ofthe accommodation response. There was noParinaud's sign. Left carotid angiograms demon-strated signs of internal hydrocephalus, and leftretrograde brachial angiography revealed anelevation of the medial posterior choroidal arterysuggesting a mass in the pineal region. lothalamate(Conray) ventriculography confirmed the presenceof hydrocephalus, including the third ventricle.

Address for reprint requests: Dr K. Higashi, Department of Neuro-surgery, Yamaguchi University School of Medicine, Ube, Yamaguchi-ken, Japan.Accepted 7 May 1979

The suprapineal recess was displaced upward andthe aqueduct was almost completely occluded.The anterior margin of the mass was clearlydelineated in the posterior part of the thirdventricle (Fig. 1). Ventricular CSF taken duringthe ventriculography was xanthochromic withprotein 0.38 g/l and 17 cells per mm3 Com-puterised tomography (CAT) scan demonstrated ahigh density mass in the pineal region, whilecontrast enhancement did not alter the densityof the mass (Fig. 2 left).The patient underwent a right occipital crani-

otomy on 10 July. The pineal region was exposedthrough the transcallosal approach with sectionof the tentorium under the microscope. On section-ing the splenium of the corpus callosum, a bluish

Fig. 1 lothalanmate (Conray) ventriculogram withair. The lateral and third ventricle are dilated. In theposterior third ventricle, there is an irregular defectwith smooth outline of the pineal mass, which pushesthe suprapineal recess upward and occludes theaqueduct almost completely.

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1051Pineal apoplexy

Fig. 2 Pre and postoperative CAT scans. Left: preoperative. Lateral ventricles aredilated. Periventricular low density is seen in front of both anterior horns. Highdensity shadow is located in the posterior third ventricle (without enhancement).Middle: 43rd postoperative day. High density shadow has completely disappeared,while ventricular size is unchanged. There is a low density area in the right occipitallobe. Right: 102nd postoperative day. Ventricular size is reduced, but low density inthe right occipital lobe remains.

coloured mass was seen, surrounded by the in-ternal cerebral vein and by Rosenthal's vein.Several fine vessels appeared to run on the surfaceof the tumour. Puncture of the tumour yieldeddark red liquid haematoma, and aspiration of

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about 4 ml of this fluid made the tumour shrink.The mass consisted of a blood clot which wasremoved piecemeal. The capsule was 2 mm thickin the posterior part and thinner in the anteriorportion. There were also blood clots outside the

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Fig. 3 Photomicrographs of the capsule of haematoma. Left: organised haematomais surrounded by gliosis with haemosiderin-laden macrophages. Haematoxylin andeosin (H and E), original magn fication X60. Right: psammoma body is seen withinvascular granulation tissue. H and E, original magnification X 150.

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capsule anteriorly, between the capsule andependyma of the third ventricle. Opening of thethird ventricle revealed that the inside of theependyma was intact. The capsule was totallyremoved.

Histological study of the capsule of the haema-toma demonstrated very vascular granulationtissue with abundant haemosiderin-laden macro-phages and hyalinized connective tissue surround-ing the organised haematoma. Proliferation ofastroglia was seen in some areas. Although therewere a few psammoma bodies, distinct pinealtissue was not observed (Fig. 3).Because of persistence of internal hydro-

cephalus, a ventriculoperitoneal shunt was placedon the tenth postoperative day, but abdominalpain with high fever began two weeks later.Suspicion of shunt infection compelled theremoval of the shunt system and symptoms dis-appeared promptly thereafter. Hydrocephalus re-lated symptoms no longer appeared after removalof the shunt, and papilloedema cleared completely.A CAT scan performed on the forty-third post-operative day revealed disappearance of the highdensity shadow in the pineal region, though thesize of the ventricles was unchanged. There was alow density area in the right occipital lobe whereit had been retracted during surgery (Fig. 2middle). The postoperative course was uneventfulexcept for the above-mentioned events and upwardgaze palsy which developed postoperatively. Thepatient was discharged on the fifty-seventh post-operative day without any neurological deficitexcept for upward gaze palsy and sluggish lightreflexes. Re-examination by CAT scan on 16October demonstrated reduction of ventricularsize, while the low density area in the rightoccipital lobe still remained (Fig. 2 right).Discussion

This patient, whose initial symptom was subarach-noid haemorrhage, had an encapsulated haema-toma located in the pineal region, which occludedthe Sylvian aqueduct and caused internal hydro-cephalus. In spite of ventriculographic demon-stration of a mass in the posterior third ventricleresembling pineal tumour, a high density shadowin the CAT scan led to the suspicion of haema-toma. As causes of intracranial haematoma inthis region, either haemorrhage into a pinealtumour (pineal apoplexy) or a vascular anomalylocated in this region should be considered.Haemorrhage into brain tumour is common

when the patient's symptoms increase suddenly.

K. Higashi, S. Katayama, and T. OritaMoreover, cerebral tumours have occasionallybeen found in patients with massive haemorrhagepresenting clinically like haemorrhagic strokes.Richardson and Einhorn (1963) reported sevensuch cases in their 108 necropsy cases of primaryintracerebral haemorrhage. Although more than180 cases of pituitary apoplexy have been re-ported (Rovit and Fein, 1972) little reference hasbeen made to pineal apoplexy.The case of Apuzzo et al. (1976) may be the

only case reported so far. On reviewing the litera-ture, they were unable to find any other cases ofhaemorrhage from a pineal tumour. Histologicalexamination of their case indicated that thehaemorrhage occurred into the pineal cyst withoutevidence of neoplasm. Among pineal tumours, thenon-neoplastic cyst is a rare entity. It is usuallyseen in adults as a residue of a pineal diverticulumor related to a degenerative process in the centralcore of the pineal gland (Russell and Rubinstein,1971).Subarachnoid haemorrhage associated with

brain tumour is uncommon. Locksley et al. (1966)found only 28 cases among 5836 patients withsubarachnoid haemorrhage. Subarachnoid haemor-rhage from pineal tumour is particularlyexceptional. Steinbock et al. (1977) recently re-ported two cases of pineocytoma presenting assubarachnoid haemorrhage, and they believed thatthese were the first examples of pineocytomaspresenting in such fashion. Our present case alsohad subarachnoid haemorrhage as an initialsymptom. In view of the operative findings, bloodclots outside the capsule may reasonably explaina subarachnoid haemorrhage secondary to ruptureof the haematoma capsule.Haemorrhage from a vascular anomaly or

vascular tumour in the pineal region is also rare.Miller (1961) reported a case with haematoma inthe posterior third ventricle caused by haemor-rhage from a cavernous haemangioma involvingthe cistern of the great cerebral vein and associ-ated recurrent subarachnoid haemorrhage.Although such an aetiology must be considered inour case, we were unable to find either an ab-normal vascular shadow in the angiograms or anyvascular anomaly on histology. On the basis ofthe operative findings and the histology of thehaematoma capsule, we concluded that this is acase of haemorrhage into a pineal cyst similar tothe case of Apuzzo et al. (1976). The haematomacapsule was totally removed by microsurgery.Nevertheless, upward gaze palsy developed post-operatively, probably because of damage topretectal tissue during surgery.

Pineal apoplexy

References

Apuzzo, M. L. J., Davey, L. M., and Manuelidis,E. E. (1976). Pineal apoplexy associated with anti-coagulant therapy. Case report. Journal of Neuro-surgery, 45, 223-226.

Araki, C., and Matsumoto, S. (1969). Statisticalreevaluation of pinealoma and related tumours inJapan. Journal of Neurosurgery, 30, 146-149.

Katsura, S., Suzuki, J., and Wada, T. (1959). Astatistical study of brain tumors in the neuro-surgical clinics in Japan. Journal of Neurosurgery,16, 570-580.

Locksley, H. B., Sahs, A. L., and Sandler, R. (1966).Report on the cooperative study of intracranialaneurysms and subarachnoid hemorrhage unrelatedto intracranial aneurysm and arteriovenous mal-formation. Journal of Neurosurgery, 24, 1034-1056.

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Miller, R. H. (1961). Spontaneous subarachnoidhemorrhage: A presenting symptom of a tumor ofthe third ventricle. Surgical Clinics of NorthAmerica, 41, 1043-1048.

Richardson, J. C., and Einhorn, R. W. (1963).Primary intracerebral hemorrhage. ClinicalNeurosurgery, 9, 114-130.

Rovit, R. L., and Fein, J. M. (1972). Pituitaryapoplexy: a review and reappraisal. Journal ofNeurosurgery, 37, 280-288.

Russell, D. S., and Rubinstein, L. J. (1971). Pathologyof Tumours of the Nervous System, third edition,pp. 217-218. Edward Arnold: London.

Steinbock, P., Dolmen, C. L., and Kaan, K. (1977).Pineocytomas presenting as subarachnoid hemor-rhage. Report of two cases. Journal of Neurosurgery,47, 776-780.