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8. Burke RE, Fahn S, Marsen CD. Validity and reliability of a rating scale for the primary torsion dystonia. Neurology 1985; 35: 73–77. 9. Beasley CM, Tollefson GD, Tran PV. Safety of olanzapine. J Clin Psychiatry 1997; 58(suppl. 10): 13–17. 10. DeLong MR, Crutcher MD, Georgopoulos AP. Primate globus pallidus and subthalamic nucleus: Functional organization. J Neurophysiol 1985; 53: 530–543. 11. Partel K, Roskrow T, Davis JS. Dopa-responsive dystonia. Arch Dis Child 1995; 73: 256–257. 12. Ichinose H, Ohye T, Takayashi E et al. Hereditary progressive dystonia with marked diurnal fluctuation caused by mutations in the GTP cyclohydrolase I gene. Nat Genet 1994; 8: 236–242. 13. Vitek JL, Chockkan V, Zhang JY et al. Neuronal activity in the basal ganglia in patients with generalized dystonia and hemiballismus. Ann Neurol 1999; 46: 22–35. 14. Wichmann T, Delaong MR, Viteck JL. Pathophysiological consideration in basal ganglia surgery: Role of the basal ganglia in hypokinetic and hyperkinetic movement disorder. In: Lozano AM (ed) Movement Disorder Surgery. Prog Neurol Surg. Karger, Basel 2000; 31–57. 15. Crossman AR. A hypothesis on the pathophysiological mechanisms that underly levodopa- or dopamine agonist-induced dyskinesia in ParkinsonÕs disease: implications for future strategies in treatment. Mov Disord 1990; 5: 100–108.
A distal anterior cerebral artery aneurysm in infant: disappearance and reappearance of the aneurysm Osamu Motohashi1 MD, Motonobu Kameyama1 Shigeki Imaizumi1 MD, Masaki Mino1 MD, Hiroshi Naganuma2 MD, Kiyoshi Ishii3 MD, Takehide Onuma1 MD
INTRODUCTION The incidence of intracranial aneurysms in childhood is very rare, especially in infancy.1;2 The incidence of intracranial aneurysms in patients under 18 years old is said to comprise only 1–2% of all aneurysms.2 The youngest patient reported harbouring an intracranial aneurysm was a 7-week-old female.3 We present a rare case of the infantile distal anterior cerebral artery (ACA) aneurysm which disappeared spontaneously after the aneurysm ruptured and reappeared 6 months later.
CASE REPORT This 1-month-old girl was referred to our hospital suffering from projectile vomiting, fever of 38.2 C and convulsions. She was born after a normal 40-week-pregnancy and an uncomplicated vaginal delivery. Until her present illness, there was no past medical history and she had no noticeable physical anomalies. On admission she was drowsy and her anterior fontanelle was tense. Computed tomography (CT) showed subarachnoid hemorrhage (SAH). The haemorrhage was mainly in the interhemispheric fissure and in the genu of the corpus callosum (Fig. 1). CT angiography (CTA) revealed an aneurysm of the right distal anterior cerebral artery (Fig. 2 left). Haematological examination indicated no inflammatory diseases. Anticonvulsants and glycerol were administrated intravenously. On day 6, while awaiting surgery, the aneurysm disappeared and the parent artery shown to be occluded on follow up CTA (Fig. 2 right). She was thus treated conservatively and on day 20, dis-
MD,
1
Department of Neurosurgery, Sendai City Hospital, Sendai, Japan, Department of Pathology, Sendai City Hospital, Sendai, Japan, 3 Department of Radiology, Sendai City Hospital, Sendai, Japan 2
Summary The incidence of intracranial aneurysms in childhood is rare, especially in infancy. Spontaneous thrombosis of a cerebral aneurysm in a child is very rare, particularly in a non-giant aneurysm. We report a case of a 1-month-old girl with a distal anterior cerebral artery aneurysm which disappeared spontaneously after subarachnoid hemorrhage and reappeared 6 months later. Surgical resection of the aneurysm was performed and she discharged uneventfully 10 days later. Histological examination revealed an aneurysm with a fibrous muscular layer, absence of the internal elastic lamina and partial hypertrophy of the intimal layer. Though the pathogenesis of this aneurysm is uncertain, two hypotheses are discussed. ª 2003 Elsevier Ltd. All rights reserved.
Fig. 1 (left) Computed tomography (CT) on admission showed the subarachnoid hemorrhage (SAH) at the basal cistern, mainly in the interhemispheric fissure. (right) Thick interhemispheric SAH and the hematoma were seen on the corpus callosum.
Journal of Clinical Neuroscience (2004) 11(1), 86–88 0967-5868/$ - see front matter ª 2003 Elsevier Ltd. All rights reserved. doi:10.1016/j.jocn.2003.09.004
Keywords: cerebral aneurysm, child, infant, thrombosis Received 29 July 2002 Accepted 12 September 2003 Correspondence to: Osamu Motohashi MD, Department of Neurosurgery, Sendai City Hospital, 3-1 Shimizukoji, Wakabayashi-ku, Sendai 984-8501, Japan. Tel.: +81-22-266-7111; Fax: +81-22-211-8972; E-mail:
[email protected]
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Fig. 2 (left) CT angiography (CTA) on admission showed the fusiform aneurysm of the right distal anterior cerebral artery. (right) CTA on the day 6 showed disappearance of the aneurysm and parent artery. No vasospasm was recognized. (upper) Antero-posterior view. (lower) Lateral view.
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charged without neurological deficit. CT scan at discharge showed no cerebral infarctions and no hydrocephalus. Follow up MRA taken 6 months later showed reappearance of the aneurysm (Fig. 3) and cerebral angiography was thus performed. An irregular shaped aneurysm was demonstrated on the right ACA with the distal ACA occluded (Fig. 4). She underwent resection of the aneurysm through a bifrontal craniotomy and interhemispheric approach. Two arterial branches arising from the aneurysm dome were sacrificed and the 7 mm aneurysm totally resected. The distal ACA appeared only to be vestigial. Pathological examination of the aneurysm revealed a thin fibrous muscular layer and partial intimal thickening with discrete internal elastic lamina (Fig. 5). She was discharged uneventfully 10 days after surgery.
Fig. 5 Photomicrographs of the aneurysm were shown. (a) Aneurysm had a thin fibrous wall (asterisk) with the partial intimal thickening (star). (b) High power field of the area in Fig. 1(a) asterisk. Fibrosis of the muscular layer was seen. The internal elastic layer was absent. (c) High power field of the area in Fig. 1(a) star. Thickening of the intima was mainly due to the proliferation of the fibroblasts producing the extracellular matrices. The internal elastic layer was discrete. (a) and (b) Hematoxylin-eosin staining. (c) Elastica Van Gieson staining. Original magnification: (a) 5, (b) 40, (c) 16.
DISCUSSION Common features of the infantile aneurysm Kasahara et al.4 and Kunimine et al.5 reviewed 43 and 31 cases of infantile intracranial aneurysms, respectively, finding certain common features in their series. Most cases were in early infancy (58–64%) and no sex difference was found. There was a high incidence (26–39%) of aneurysms occurring in the posterior circulation. Giant aneurysms were most frequent and the most common presentation was SAH due to aneurysmal rupture. Fig. 3 MRA after 6 months showed reappearance of the aneurysm. The shape of the aneurysm changed compared to that at initial CTA.
Aneurysms of the distal ACA in infancy The presence of aneurysms of the distal ACA in infancy is quite rare.3 We were able to find only six reported cases of distal ACA or pericallosal artery aneurysms in the literature,3;6–10 with detailed clinical information available in only five cases. These five patients all presented with haemorrhage, a decreased level of consciousness and seizures as their initial symptoms. Agenesis of the corpus callosum was recognized in one case.9 Five of the six cases underwent craniotomy and clipping with varying outcomes. Of the five patients who underwent surgery, the outcome in two was favorable, two had fair outcomes and the outcome in the fifth was not specified. The patients treated conservatively died.9 The aetiology in two cases was attributed to trauma,6;7 two cases were congenital9;10 and the aetiology in the last two was not specified.3;11 Detailed histological examination was performed in two cases.6;9 The internal elastic membrane was abnormal in both these cases, despite having a different pathogenesis.6;9 Pathogenesis of the pediatric intracranial aneurysms
Fig. 4 Right internal carotid angiography showed the irregular shaped aneurysm on the right anterior cerebral artery which was occluded distal to the aneurysm. (upper left) Antero-posterior view. (upper right) Lateral view. (lower left) Right anterior oblique view. (lower right) Left anterior oblique view.
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Norris and Wallace2 reported that up to 55% of paediatric patients with aneurysms have an underlying disorder or atypical characteristic associated with aneurysm formation. In childhood and adolescence, secondary hypertension due to coarctation of the aorta nor polycystic kidney disease has been emphasized as an important factor in aneurysm formation.5 In cases of aneurysms in infancy none had coarctation of the aorta nor hypertension.5 Kasahara et al.4 suggested birth trauma in addition to a congenital origin as a possible aetiology of infantile aneurysms. Several Journal of Clinical Neuroscience (2004) 11(1)
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authors pointed out that the anatomical relationship between the artery and the dural edge, that is the falx or tentorium, possibly contributed to the pathogenesis of the aneurysms.12;13 Norris and Wallace suggested that the congenital factors might play a limited role in the aneurysm formation in infancy based on the report of angiography and histological studies.2;14;15 Spontaneous thrombosis of the cerebral aneurysm Spontaneous thrombosis of a cerebral aneurysm in a child is rare, particularly of a non-giant aneurysm.16 Nanda and Drury16 reviewed 28 cases of spontaneously thrombosed aneurysms and stated that the tendency of giant aneurysms to spontaneously thrombose is related to the size ratio of the aneurysm to its neck, haemodynamic considerations and the age of the aneurysm. The role of SAH in spontaneous thrombosis is controversial.16 Spetzler et al.17 reported an adult case of an aneurysm that repeatedly disappeared and reappeared on serial angiography. Though they hesitated to present a conclusion on the pathogenesis of this phenomenon, they did refer to the possible effect of the e-aminocaproic acid or of marked vasospasm. In our case, we were uncertain as to the pathogenesis of the aneurysm, but considered two possibilities namely congenital and acquired. The former may be unlikely in view of the angiography findings and histological studies suggesting that congenital factors played a limited role in aneurysm formation in infancy.2;14;15 The possibility of birth trauma in the pathogenesis is more tempting when considering the varying aneurysmal shapes. This suggests dissection and because of the falx constantly shifting its position to the distal ACA during delivery. The absence of fresh dissection on histological examination is not incompatible with the diagnosis of dissection as the specimen was taken 6 months after presentation.18 The mass effect of the haematoma around the aneurysm and delayed vasospasm might play some role in the transient spontaneous disappearance of the aneurysm and the parent artery occlusion. Another possibility was that disappearance of the aneurysm and the parent artery was the result of the arterial dissection.
11. Milhorat TH. Pediatric Neurosurgery. FA Davis Co, Philadelphia 1978. 12. Yazabak PA, McComb JG, Raffel C. Pediatric traumatic intracranial aneurysms. Pediatr Neurosurg 1995; 22: 15–19. 13. Meyer FB, Sundt Jr TM, Fode NC, Morgan MK, Forbes GS, Mellinger JF. Cerebral aneurysms in childhood and adolescence. J Neurosurg 1989; 70: 420–425. 14. Harwood-Nash DC. Paediatr Radiol 1972; 10: 313–331. 15. Stehbens WE. Intracranial berry aneurysms in infancy [review]. Surg Neurol 1982; 18: 58–60. 16. Nanda A, Drury BT. Posterior circulation aneurysms in a child: clipping of one leads to spontaneous thrombosis of the other. Pediatr Neurosurg 1997; 26: 41–47. 17. Speztler RF, Winestock D, Newton HT, Boldrey EB. Disappearance and reappearance of cerebral aneurysm in serial arteriograms. Case report. J Neurosurg 1974; 41: 508–510. 18. Nakatomi H, Nagata K, Kawamoto S. Persistent patent pseudolumen of ruptured dissecting aneurysm involving the posterior inferior cerebellar artery after proximal clipping. Acta Neurochir 1999; 141: 533–536.
Single choroid plexus metastasis 16 years after nephrectomy for renal cell carcinoma: case report and review of the literature David Kadrian
B.MED,
Leong Tan
MBBS
Department of Neurosurgery, Gold Coast Hospital, Southport, Qld 4215, Australia
Journal of Clinical Neuroscience (2004) 11(1), 88–91 0967-5868/$ - see front matter ª 2003 Elsevier Ltd. All rights reserved. doi:10.1016/j.jocn.2002.09.002
Received 20 August 2002 Accepted 19 September 2002
ACKNOWLEDGEMENTS The authors thank Haruki Murayama for providing technical assistance.
Correspondence to: David Kadrian, Department of Neurosurgery, Gold Coast Hospital, Southport, Queensland, Australia.
REFERENCES
INTRODUCTION
1. Hulsmann S, Moskopp D, Wassman H. Management of a ruptured cerebral aneurysm in infancy – report of a case of a ten-month-old boy. Neurosurg Rev 1998; 21: 161–166. 2. Norris JS, Wallance MC. Pediatric intracranial aneurysms. Neurosurg Clin N Am 1998; 9: 557–563. 3. Frank E, Zusman E. Aneurysms of the distal anterior cerebral artery in infants. Pediatr Neurosurg 1990; 16: 179–182. 4. Kasahara E, Murayama T, Yamane C. Giant cerebral arterial aneurysm in an infant: report of a case and review of 42 previous cases in infants with cerebral arterial aneurysm. Acta Paediatr Jpn 1996; 38: 684–688. 5. Kunimine H, Inoue H, Isobe I, Nukui H. Case of intracranial aneurysm in an infant: together with evaluation of 31 cases from the literature. Neurol Surg 1983; 11: 531–538. 6. Lam CH, Montes J, Farmer JP, OÕGorman AM, Meagher-Villemure K. Traumatic aneurysm from shaken baby syndrome: case report. Neurosurgery 1996; 39: 1252–1255. 7. Amacher AL, Drake CG. Cerebral artery aneurysms in infancy, childhood and adolescence. ChildÕs Brain 1975; 1: 72–80. 8. Ferry PC, Kerber C, Perterson D, Gallo AA. Arteriectasis subarachnoid hemorrhage in a three-month-old infant. Neurology 1974; 24: 494–500. 9. Garcia-Chavez C, Moossy J. Cerebral artery aneurysm in infancy. Association with agenesis of the corpus callosum. J Neuropathol Exp Neurol 1965; 24: 492–501. 10. Morelli RJ, Laubscher F. Intracranial aneurysm in infancy. Case report. J Neurosurg 1977; 46: 832–834.
Amongst intraventricular tumours, those within the choroid plexus represent a small subset, around 7%.16 Of these, choroid plexus papillomas, metastatic lesions of primarily parenchymal origin with secondary extension into the choroid plexus, and the less common choroid plexus carcinomas account for most tumours.16;17;26 Particularly rare are solitary metastases to the choroid plexus from extra-neural primary sites. Only 18 cases have been reported so far (Table 1). Of these 18 cases, 8 arose from renal cell carcinoma. While renal cell carcinoma metastasises to the brain in anywhere from 5% to 10% of cases,1;5;8;18;23 solitary metastasis to brain is much less common (0.6–2.5% in large autopsy series).8;23 In this context then, solitary metastasis specifically to the choroid plexus is clearly vanishingly rare (only 8 cases reported, Table 1). Furthermore, while solitary brain metastasis of renal cell carcinoma has been reported with latency periods of more than 10 years post nephrectomy,8;12;20;22 there are no such long latency periods reported for solitary metastasis to the choroid plexus. One of the 8 reported cases was identified 4 years after the diagnosis of renal cell carcinoma10 and another 7 years after left nephrectomy for renal cell carcinoma.13
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