Acta Ncurochirurgica 81, 72-76 (1986) :Acta . . Nurochlrurglca 9 by Springer-Verlag 1986 Aneurysm Inside Meningioma C a s e Report E. Kandel, I. Ludkovskaya, and N. Dobjansky Neurosurgical Clinic, Institute of Neurology, Moscow, USSR Summary The first case in the literature of a large meningioma of the Sylvian fissure incorporating an aneurysm of the middle cerebral artery in a 7-year-old girl is described. The tumour with the aneurysm inside was totally removed. Despite resection of the middle cerebral artery there was complete disappearance of a severe pre-operative neurological deficit. The patient is practically healthy six years after surgery. Keywords: Meningioma; arterial aneurysm; middle cerebral artery. We present the unusual case of an aneurysm of the middle cerebral artery inside a large meningioma. Case Report A 7-year-old girl was admitted to our clinic in July 1977. When the child was 2 years old the first short syncope-like petit mal seizure took place and then recurred once or twice a year. There were no other complaints, no neurological symptoms or signs. One week before admission, motor unrest and after that a deep right-sided hemiparesis and semicomatous state suddenly and quickly developed. A lumbar puncture at a local hospital revealed grossly bloody CSF. The following day hemiplegia and total aphasia with severe meningeal signs were noted. When admitted the girl was in a grave condition. She was confused and inactive. There were no movements in her right arm and minimal movements in the right leg with pathological reflexes, severe aphasia, hemihypaesthesia, initial signs of optic disc oedema. The Mecho showed a 5 mm shift to the right. Carotid angiography disclosed a typical arterial aneurysm of the left middle cerebral artery (size 7 x 6 mm). The artery was displaced medially and the anterior cerebral artery to the right (Figs. 1 a and b). CT-scans showed a large (6 x 6 cm) lesion of high density with irregular but distinct contours in the left fronto-temporal region surrounded by a narrow oedematous zone. Inside the lesion, an area of accumulated contrast medium was noted (Fig. 2). According to the above-mentioneddata, the clinical suggestion of a massive parenchymatous haematoma after aneurysm rupture was expressed. Operation. A typical left fronto-temporal craniotomy was performed under general anaesthesia. A mild oedema was noted. In the Sylvian fissure I cm under the Cortex a very large ball-shaped tumourlike mass was found, which replaced the anterior half of the temporal and the posterior part of the frontal lobe. This encapsulated mass was carefully separated from the normal brain tissue. The proximal part of the middle cerebral artery (M1) deep in the Sylvian fissure entered into the inferior pole of the mass, while the distal part of the artery left its superior pole. It was suggested that it was a giant arterial aneurysm partly thrombosed and filled with a clot, which may explain the small size of the aneurysm on the angiograms. Because of the impossibility of separating the mass and the middle cerebral artery inside it, both ends of the artery were ligated and cut. The diastase between the two ends was about 5 cm. After that, the mass was totally removed en bloc; it was about 6 cm in diameter. The inferior pole of the mass contacted the bottom of the middle cranial fossa, where a relatively small haematoma (about 15 ml) was found and removed. During the early post-operative course an uncommonly rapid recovery was noted. The movements in the paralyzed extremities and spontaneous speech re-appeared on the 3rd day and improved rapidly. On the 12th day the girl began to walk. Three weeks after the operation she was discharged with only light hemiparesis and mild amnestic aphasia. 6 years after the operation the girl is practically healthy and has no complaints (Fig. 3). There are no neurological signs exept light asymmetry of reflexes in the legs. Anatomical findings: MacroscopicalIy, the cross-section of the removed ball-shaped mass of elastic consistency consisted of a thick peripherial part of grey-rose tissue and the centre replaced by a haematoma. Inside the haematoma an arterial aneurysm about 6 mm in diameter surrounded by coagulated blood and parts of the middle cerebral artery were disclosed (Fig. 4). Microscopically, the peripheral tissue has the structure of a typical fibroblastic meningioma (Fig. 5) with the compact linings of cells, containing oval nuclei filled with chromatin. The cell linings present concentric structures (Fig. 6). The artery in the central part of the tumour was 1.5mm in diameter and had a typical three-layered wall. The wall of the E. Kandel et al.: Aneurysm Inside Meningioma 73 Fig. 1. Pre-operative carotid angiography, a) lateral, b) A-P angiograms. Note the aneurysm of the left middle cerebral artery. The anterior cerebral artery is displaced to the right Fig. 2. CT with contrast enhancement before the operation. Note the big zone o fincreased density in the left fronto-temporal region. In the central part of the zone there is a round lesion of contrast uptake. The zone is surrounded by a strip of low density (brain tissue oedema). The middle structures and anterior horns are markedly displaced to the right. Posterior horn and third ventricle are compressed aneurysm consists of collagen fibres onIy. In the neck of the aneurysm typical changes were lbund such as: absence of the internal elastic membrane, defects of the muscular layer. There was a zone of fibrinoid necrosis in the aneurysm wall, surrounded by fresh thrombotic masses. It was the site of the aneurysm rupture and bleeding into the tumoral tissue. In some places the adventitia of the artery was intimately attached Fig. 3. The girl is healthy five years after surgery to the tumoural tissue (Fig. 7). It may be suggested that the arachnoid surrounding the artery was the source of tumoral growth. Discussion According to the literature an association of an aneurysm with different types of tumours in the same 74 E. Kandel etal.: Aneurysm Inside Meningioma Fig. 4. Large meningioma (big arrow) with haematoma inside (small arrow). Note berry-shaped aneurysm in the centre of the haematoma (two arrows). The aneurysmal wall is in the state of fibrinoid necrosis. The middle cerebral artery has the typical structure (arrowhead). H. E. Microplanar x 5 Fig. 5. Microscopic picture of fibroblastic meningioma. H. E. x 200 brain occur very rarely. The rate o f such coexistence is 0.2-0.7% o f all brain t u m o u r cases 3, 4, 10, 15. In several reports only 1-2 cases were described 1, 2, 5-9, 11-14 In 1961 T a y l o r 15 f o u n d 7 published cases in the literature o f the previous 65 years and 8 unreported cases f r o m English neurosurgical clinics with the addition o f one personal case. In 1972 Pia, O b r a d o r and M a r t i n 10 have analyzed 116 cases o f the association collected f r o m the literature and by questionnaires f r o m m a n y leading clinics. The authors added eight personal cases f r o m their clinics in Giessen and Madrid. In the above-mentioned coexistence the initial signs and s y m p t o m s were connected with the t u m o u r in 66% and with the a n e u r y s m in 22% o f the cases. Only 34 brain t u m o u r s f r o m 116 cases collected by E. Kandel et al.: Aneurysm Inside Meningioma 75 Fig. 6. Concentric structures in the tissue of the meningioma. H.E. x 200 Fig. 7. The growth of the tumour cells from the adventitia of the middle cerebral artery to the tumoural tissue. H,E, x200 the authors were meningiomas. It is noteworthy that the incidence of the coexisting aneurysms with meningiomas and pituitary adenomas is about two times higher than with other brain tumours. The higher relative frequency of middle cerebral aneurysms, especially with convexity tumours was also stressed. There was a predominance of aneurysms ipsilateral to the tumour and related to the arteries which supply the tumour I0 In 1976 H a n d a et al. collected from the literature 134 cases of intracranial aneurysms occurring with brain tumours and added seven personal cases. G6kalp e t al. 3 described four such personal cases. Nevertheless, we could not find in the literature any case of the location of an aneurysm inside a meningioma as in our case. The surgical treatment of coexisting aneurysm and brain tumour is connected with a high risk irrespective of whether the tumour, the aneurysm, or both are 76 surgically treated. A c c o r d i n g to Pia e t a l . 10 the posto p e r a t i v e m o r t a l i t y was 4 0 % . T h e genesis o f a n e u r y s m a n d t u m o u r coexistence is a m o s t interesting a n d u n s o l v e d p r o b l e m . Several o p i n i o n s were expressed in the literature. D y s g e n e t i c defects in the arterial wall a n d also in the b r a i n a n d its covering cells is the m o s t c o m m o n suggestion. It is s u p p o r t e d b y o u r case, b e c a u s e the first clinical sign (petit m a l a t t a c k s ) as early as at the age o f two years, led us to s u p p o s e a c o n g e n i t a l origin o f the t u m o u r . A c o n g e n i t a l defect in the arterial wall exactly in the same b r a i n zone m a y lead to the a n e u r y s m f o r m a t i o n . T h e o p i n i o n was expressed t h a t certain b r a i n t u m o u r s , m a i n l y o f the skull base c a u s i n g a local d i s t u r b a n c e s in c e r e b r a l circulation, m a y s e r v e as a p r e d i s p o s i n g f a c t o r for the d e v e l o p e m e n t o f a r t e r i a l a n e u r y s m s 10. I n c r e a s e d r e g i o n a l b l o o d flow because o f t u m o u r s u p p l y m a y also p l a y a role in a n e u r y s m f o r m a t i o n 3, 10. T h e a b o v e - m e n t i o n e d p r e d o m i n a n t ipsilateral l o c a t i o n o f a n a n e u r y s m a n d a t u m o u r a n d its r e l a t i o n to the s a m e a r t e r y s u p p o r t this suggestion. T h e r e was a n o p i n i o n t h a t direct t u m o u r influence on the a r t e r i a l wall m a y also be involved. I n o u r case, the m e n i n g i o m a m a y be suggested to have d a m a g e d the m i d d l e c e r e b r a l a r t e r y wall acting as a n aetiological f a c t o r in a n e u r y s m a l f o r m a t i o n . T h e close a t t a c h m e n t o f t u m o u r a l tissue to the arterial wall, p r o v e d in o u r case m i c r o s c o p i c a l l y , m a y s u p p o r t the suggestion. But a c o u n t e r - a r g u m e n t m a y be p r o p o s e d : m e n i n g i o m a s quite often include m a j o r c e r e b r a l vessels w i t h o u t such consequences. It seems c o n s i d e r a b l y less p r o b a b l e t h a t the m e n i n g i o m a d u r i n g its g r o w t h a r o u n d the m i d d l e c e r e b r a l a r t e r y has i n c l u d e d a c o n g e n i t a l a n e u r y s m o f the artery. E. Kandel et al.: Aneurysm Inside Meningioma 2. 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Arseni C, Maretsis M (1973) Meningioma associated with intracranial aneurysm or cerebral thrombosis. Neurochirurgia (Stuttg) 16:131-137 Authors' address: Prof. E. Kandel, Neurosurgical Clinic, Institute of Neurology, Moscow 123367, USSR.