ELSEVIER Brain & Development 1994; 16:233-7 Case Report A case of cerebral aneurysm associated with complex partial seizures Katsuji Tanaka a, MD, Ken Hirayama a, MD, PhD, Hideji Hattori a MD, PhD, Osamu Matsuoka a, MD, Hiroaki Sakamoto b, MD, PhD, Akira Hakuba b, MD, PhD, Ryosuke Murata a, MD, PhD We report a 20-month-old girl with an unruptured aneurysm of the middle cerebral artery. The initial sign was complex partial seizures. Magnetic resonance angiography showed an aneurysm of the right middle cerebral artery. Cerebral angiography confirmed the presence of a saccular aneurysm. After 3 months, the aneurysm spontaneously thrombosed without neurological deficit. The seizures were controlled by anti-convulsants. The aneurysm was probably near the focus of the complex partial seizures. This is a rare case of a childhood cerebral artery aneurysm that first manifested itself by complex partial seizures. Key words." Cerebral aneurysm; Cerebral vascular anomaly; Epileptogenic focus; Complex partial seizure in infancy; Magnetic resonance angiography 1. INTRODUCTION Cerebral aneurysms are rare in early childhood [1]. One common result of an aneurysm in both adults and children is subarachnoid hemorrhage, and epilepsy from this cause is rare. We report a patient with an unruptured aneurysm of the right middle cerebral artery (MCA) who presented with complex partial seizures. The aneurysm was detected by magnetic resonance angiography (MRA), and it thrombosed spontaneously, which is rare in childhood [2]. Departmentsof a Pediatrics and b Neurosurgery, Osaka City University Medical School, Osaka, Japan Received 7 October 1993; accepted 16 January 1994 Correspondence address: Dr. K. Tanaka, Department of Pediatrics, Osaka City University Medical School, 1-5-7 Asahi-machi, Abeno-ku, Osaka 545, Japan. Fax: (81) (6) 646-5862. 0387-7604/94/$07.00 © 1994 ElsevierScience B.V. Allrightsresewed SSDI 0 3 8 7 - 7 6 0 4 ( 9 4 ) 0 0 0 1 7 - R 2. CASE REPORT A 20-month-old girl was hospitalized at our hospital for treatment of seizures. The first episode, in which the patient suddenly stared to the left side for about 1 min with impairment of consciousness, occurred when she was 16 months old. The seizures reoccurred every few days. She was first hospitalized elsewhere. Results of electroencephalography ( E E G ) are shown in Fig. 1. There were spikes and slow waves in the right occipital area (Fig. 1A). Computed tomography (CT) of the brain revealed a high-density lesion in the right temporal lobe (Fig. 2A). Complex partial seizures were diagnosed from the E E G findings and seizure type. Phenytoin and valproate did not control the seizures. The patient was referred to our hospital for further evaluation and therapy. The family history and perinatal history were negative. Development was slightly delayed; she first walked alone at the age of 18 months. There were no other neurological abnormalities. The IL Tanaka et al. /Brain & Decelopment 1994, 16:233 7 234 A B Fp+-Ai C3-P3 " . . . . " +.+ < +" "+.p~" . ' ,~:.+.m"' ' & +," .: ".+?'., .... , ,, + . . . F7-Fp'~ . .]v~ C3-AI ". . +.. O1-T3 #"f.,,"~ ,"+- ~,¢+,." " + . . Fp2-A2 P~-O~ ,+, . C . Fp~..F8 , ,.. ,: , ' C4-A2 " ~" "~+'+,,+~" ' ~+ % t c ~ V +~v-.'~ V + '%: + ~ : ""+ F7-C3 ., , . ,$' .:. ,%, N A T3,-F7 FT-Fpl T3-P3 Fpz,-C4 O2,-A2 " C4-P4 . .+ ,~. '. FT-AI ~', P4-T4 "~+'%~"'.:'- ~ +~'~<.:'~'++~/-X~'~J"]'J:'~ --" P4-C>2 T3-.01 .++,,~.:.:,jL-%~,;'~+.,'..+~,:q-,,~£~.,/'( " ", ,.+'" O2-T4 v T4,-Fe "" 01-02 +:"~.+"" ,+ : J . . : - ~ % - v - + . . " + ' + + : ~ ' - % . + ~ ^ 'J ' d " -.' ....." %"+"'° . . . . " T+-A2 Fe-Fp2 , ~ j , , + , ,-- + i j i y :w:+. ECG I J ECG Fig. 1. A : temporal EEG at 16 months of age. T h e spikes and waves are in the right occipital area. B,C: EEG on admission. The sharp wave is in the right area and the spikes are in the right central area. laboratory data were normal. EEG showed focal spikes and sharp waves in the right central and temporal areas (Fig. 1B,C). Contrast-enhanced CT showed enhancement of the lesion (Fig. 2B). T:weighted magnetic resonance imaging (MRI) showed a high-signal area surrounding a signal void in the right hippocampus (Fig. 3A). T:weighted images showed a signal void with a high-signal area inside (Fig. 3B). Because these findings suggested that there was a vascular disorder, probably a partially thrombosed aneurysm, MRA was Fig. 2. A: plain C T scan at 16 months of age. T h e r e is a high-density area in the right temporal lobe. B: contrast-enhanced C T scan at 20 months. The right temporal lesion is enhanced. K. Tanaka et al. /Brain & Development 1994; 16." 233-7 done. M R A showed a n a n e u r y s m in the M~ p o r t i o n of the right M C A (Fig. 3C). This a n e u r y s m was also revealed by c e r e b r a l angiography, which showed a sac- 235 cular a n e u r y s m of the right M C A m e a s u r i n g 15 × 8 m m (Fig. 3D). T h e r e was stenosis of the right M C A proximal to the a n e u r y s m . Fig. 3. Initial MRI, cerebral angiography, and MRA. A: the Tl-weighted image shows a signal void surrounded by a high-signal area in the right hippocampus. B: the T2-weighted image shows a signal void containing a smaller high-signal area. C: MRA shows a large aneurysm in the M~ portion of the right MCA. D: digital subtraction angiography done during the first admission shows a large saccular aneurysm of the right MCA (arrow). The parent artery is narrow. 23~ K. Tanaka et al. / Brain & Del~elopment 1994," 16." 233-7 The seizures were controlled by valproate, diazepam, and carbamazepine. Three months later, the patient was re-admitted because surgical treatment was planned. MR1 and M R A were done again. The thrombosed aneurysm was visible, but the right M C A was not (Fig. 4 A - C ) . Cerebral angiography (Fig. 4D) showed i!!!i~ili~il~ ~ ~!~,- i~i~~ %ii ¸ .D Fig. 4. MRI, MRA, and cerebral angiography on second admission. A: the Ti-weighted image shows a high-signal area in the right hippocampal region. B: the T2-weighted image shows a signal void containing a smaller high-signal area. C: MRA shows that the aneurysm had thrombosed, but that the right MCA bad not. D: digital subtraction angiography. The trunk of the right MCA and the aneurysm are not visible. K. Tanaka et al. /Brain & Development 1994; 16. 233-7 occlusion of the right MCA; the aneurysm was not seen. Through collateral vessels, there was retrograde filling of the area distal to the MCA occlusion. [123I]Iodoamphetamine single-photon emission tomography showed decreased cerebral blood flow in the right temporoparietal region. There was no neurologic deficit. 3. DISCUSSION The MCA is frequently the affected artery in children with cerebral aneurysm [1]. The signs and symptoms of unruptured aneurysms can include headache, cranial nerve palsies, and other neurologic deficits. Epilepsy is an initial manifestation of unruptured aneurysms in adults [3-6], but to the best of our knowledge, there are few reports of epilepsy in this condition in early childhood [1]. Aneurysms associated with epilepsy are usually large, and are often of the MCA [3-6]. The seizure type is usually of complex partial seizures [3-6], although the type depends on the location of the aneurysm. In this case, it seemed that the aneurysm caused the epilepsy. Complex partial seizures are uncommon in children under 3 years of age [7,8]. Etiological factors associated with complex partial seizures are birth asphyxia, congenital infections, neoplasms, arteriovenous malformations, and congenital cerebral anomalies. Aneurysm is a very rare cause of complex partial seizures [9]. It is not known how an aneurysm causes epilepsy. At least three mechanisms have been proposed: compression of the adjacent cortex by the aneurysm [4], subclinical hemorrhage from the aneurysm resulting in tissue damage with later development of an epileptic focus [4-6], and ischemia secondary to embolization by the thrombus from an unruptured thrombosed aneurysm [6]. Our patient had complex partial seizures and the E E G findings were of focal spikes and slow waves from the occipital to the temporal area in the right hemisphere. The aneurysm was adjacent to the cortex of the right medial temporal lobe, and it was large. These factors may have been associated with the seizures. During the first admission, cerebral angiography showed stenosis of the right MCA proximal to the partially thrombosed aneurysm. Occlusion of the par- 237 ent artery of an aneurysm is very rare [10]. That the patient had no neurological deficit suggests that the occlusion progressed slowly. Compression of the parent artery by the aneurysm itself and the propagation of thrombosis from the aneurysm may occlude the parent artery [10]. We assume that compression of the parent artery by the aneurysm slowed the blood flow, and that the thrombus was propagated from the thrombosed aneurysm into the parent artery. Treatment for epilepsy caused by an unruptured aneurysm includes anti-convulsant therapy and surgery. Surgery can prevent subarachnoid hemorrhage and alleviate seizures. For epilepsy, an aneurysmectomy is more effective than wrapping of the aneurysmal walls [4]. In our patient, there was little risk of subarachnoid hemorrhage, because the aneurysm and parent artery had thrombosed. REFERENCES 1. Ferrante L, Fortuna A, Celli P, Santoro A, Fraioli B. Intracranial arterial aneurysms in early childhood. Surg Neurol 1988; 29: 39-56. 2. Tanabe M, Inoue Y, Hori T. 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