小児もやもや病の術後長期成績 当科における20年間の経験から

もやもや病(ウイリス動脈輪閉塞症)は疾患概念の確立から約30年が経過し20), その病因, 病態, 治療法などが年々明らかとなりつつある1). 本疾患は小児にも好発し脳虚血発作を起こすという特徴を有しているが, さまざまな脳血行再建術が提唱されているものの, それらがもたらす5-10年以上にわたる長期予後に関しての報告は, 現在でもけっして多いとはいえない7)8)10)16)17)18). 当科では本疾患に対する脳血行再建術が始められて約20年が経過するが,今回, これまでに経験した症例の長期予後について, 初発症状, 手術時期, 手術方法, 脳虚血発作の推移, 脳循環動態, 知能, 就学およ...

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Published in脳卒中の外科 Vol. 28; no. 6; pp. 421 - 426
Main Authors 高橋, 明弘, 石川, 達哉, 中川, 翼, 阿部, 弘, 上山, 博康, 宝金, 清博, 黒田, 敏
Format Journal Article
LanguageJapanese
Published 一般社団法人 日本脳卒中の外科学会 2000
日本脳卒中の外科学会
Subjects
Online AccessGet full text
ISSN0914-5508
1880-4683
DOI10.2335/scs1987.28.6_421

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Abstract もやもや病(ウイリス動脈輪閉塞症)は疾患概念の確立から約30年が経過し20), その病因, 病態, 治療法などが年々明らかとなりつつある1). 本疾患は小児にも好発し脳虚血発作を起こすという特徴を有しているが, さまざまな脳血行再建術が提唱されているものの, それらがもたらす5-10年以上にわたる長期予後に関しての報告は, 現在でもけっして多いとはいえない7)8)10)16)17)18). 当科では本疾患に対する脳血行再建術が始められて約20年が経過するが,今回, これまでに経験した症例の長期予後について, 初発症状, 手術時期, 手術方法, 脳虚血発作の推移, 脳循環動態, 知能, 就学および就職状況など幅広い点から検討したので報告する. 対象 過去20年間に当科で経験した14歳未満で発症した小児もやもや病51例である. 男児21例, 女児30例であり, 発症は0-13歳であった. 1例を除いた50症例95半球に対して, 1-29歳時に脳血行再建術がなされた. 基本的に脳血行再建術は両側に対して実施したが, 非発作側で血管病変がきわめて軽度な症例(鈴木分類第1-2期)では片側のみの手術が行われた.
AbstractList We assessed long-term outcome after surgical revascularization in childhood moyamoya disease. During the past 20 years, 51 children with moyamoya disease were treated in our hospital. All but 1 child underwent surgical revascularization, either indirect bypass surgery or STA-MCA anastomosis combined with indirect bypass surgery. The mean follow-up period was 9.7 years. Surgical revascularization remarkably reduced or completely resolved ischemic attacks, although postoperative incidences of TIA or headache were significantly higher in children who underwent indirect bypass surgery than in those who underwent STA-MCA anastomosis combined with indirect bypass surgery. Postoperative SPECT studies also showed that STA-MCA anastomosis combined with indirect surgery improved hemodynamics in the frontal lobe. The children with TIA had a better intellectual outcome than those with cerebral infarction. If children with TIA underwent surgical revascularization soon after onset, they were able to pursue higher education and careers. Intracranial bleeding has not been observed in any subject during follow-up periods. These results suggest that surgical revascularization can improve long-term outcome in childhood moyamoya disease if the patients undergo the procedure soon after onset.
もやもや病(ウイリス動脈輪閉塞症)は疾患概念の確立から約30年が経過し20), その病因, 病態, 治療法などが年々明らかとなりつつある1). 本疾患は小児にも好発し脳虚血発作を起こすという特徴を有しているが, さまざまな脳血行再建術が提唱されているものの, それらがもたらす5-10年以上にわたる長期予後に関しての報告は, 現在でもけっして多いとはいえない7)8)10)16)17)18). 当科では本疾患に対する脳血行再建術が始められて約20年が経過するが,今回, これまでに経験した症例の長期予後について, 初発症状, 手術時期, 手術方法, 脳虚血発作の推移, 脳循環動態, 知能, 就学および就職状況など幅広い点から検討したので報告する. 対象 過去20年間に当科で経験した14歳未満で発症した小児もやもや病51例である. 男児21例, 女児30例であり, 発症は0-13歳であった. 1例を除いた50症例95半球に対して, 1-29歳時に脳血行再建術がなされた. 基本的に脳血行再建術は両側に対して実施したが, 非発作側で血管病変がきわめて軽度な症例(鈴木分類第1-2期)では片側のみの手術が行われた.
Author 宝金, 清博
高橋, 明弘
上山, 博康
石川, 達哉
阿部, 弘
中川, 翼
黒田, 敏
Author_FL KURODA Satoshi
HOUKIN Kiyohiro
ISHIKAWA Tatsuya
NAKAGAWA Yoku
KAMIYAMA Hiroyasu
ABE Hiroshi
TAKAHASHI Akihiro
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References 1) Fukui M : Current state of study on moyamoya disease in Japan. Surg Neurol 47 : 138-143, 1997
23) 吉井与志彦 : Moya-moyaの予後に関する研究 (I)-臨床予後について-. 脳神経29 : 421-424, 1977
13) Kuroda S, Kamiyama H, Houkin K, et al : Cerebral hemodynamics and“re-build up”phenomenon on electroen-cephalogram in children with moyamoya disease. Childs Nery Syst 11 : 214-219, 1995
22) Touho H, Karasawa J, Ohnishi H : Haemodynamic evaluation of parapretic transient ischaemic attacks in childhood movamova disease. Neurol Res 17 : 162-168, 1995
9) Komiyama M, Yasui T, Kitano S, et al : Moyamoya disease and pregnancy : case report and review of the literature. Neurosurgery 43 : 360-369, 1998
17) Matsushima Y, Aoyagi M, Nariai T, et al : Long-term intelligence outcome of post-encephalo-duro-arterio-synangiosis childhood moyamoya disease. Clin Neurol Neurosurg 99 (Suppl 2) : S147-S150, 1997
18) Nakashima H, Meguro T, Kawada S, et al : Long-term results of surgically treated moyamoya disease. Clin Neurol Neurosurg 99 (Suppl 2) : S156-S161, 1997
19) Sato H, Sato N, Tamaki N, et al : Chronic low-perfusion state in children with moyamoya disease following revascularization. Childs Nery Syst 6 : 166-171, 1990
20) Suzuki J, Takaku A : Cerebrovascular “moyamoya” disease : disease showing abnormal net-like vessels in the base of brain. Arch Neurol 20 : 288-299, 1969
14) Kuroda S, Houkin K, Kamiyama H, et al : Regional cerebral hemodynamics in childhood moyamoya disease. Childs Nery Syst 11 : 584-590, 1995
6) Imaizumi T, Hayashi K, Saito K, et al : Long-term outcomes of pediatric moyamoya disease monitored to adulthood. Pediatr Neurol 18 : 321-325, 1998
21) Takahashi A, Kamiyama H, Houkin K et al : Surgical treatment of childhood moyamoya disease-comparison of reconstructive surgery centered on the frontal region and the parietal region-. Neurol Med Chir (Tokyo) 35 : 231-237, 1995
2) Fukuyama Y, Umezu R : Clinical and cerebral angiographic evaluations of idiopathic progressive occlusive disease of the circle of Willis ('moyamoya'disease) in children. Brain Dev 7 : 21-37, 1985
7) Ishikawa T, Houkin K, Kamiyama H, et al : Effects of surgical revascularization on outcome of patients with pediatric moyamoya disease. Stroke 28 : 1170-1173, 1997
16) Matsushima T, Inoue TK, Suzuki SO et al : Surgical techniques and the results of a fronto-temporo-parietal combined indirect bypass procedure for children with moyamoya disease : a comparison with the results of encephalo-duro-arterio-synangiosis alone. Clin Neurol Neuro-surg 99 (Suppl 2) : S123-S127, 1997
11) Kuroda S, Kamiyama H, Abe H, et al : Acetazolamide test in detecting reduced cerebral perfusion reserve and predicting long-term prognosis in patients with internal carotid artery occlusion. Neurosurgery 32 : 912-919, 1993
15) Kurokawa T, Tomita S, Ueda K, et al : Prognosis of occlusive disease of the circle of Willis (moyamoya disease) in children. Pediatr Neurol 1 : 274-277, 1985
4) 宝金清博, 石川達哉, 高橋明弘, ほか : 小児もやもや病外科治療上の問題点-残された問題は何か?-. 脳卒中の外科26 : 38-44, 1998
8) Karasawa J, Touho H, Ohnishi H, et al : Long-term followup study after extracranial-intracranial bypass surgery for anterior circulation ischemia in childhood moyamoya disease. J Neurosurg 77 : 84-89, 1992
10) Kinugasa K, Mandai S, Kamata I, et al : Surgical treatment of moyamoya disease : operative technique for encephaloduro-arterio-myo-synangiosis, its follow-up, clinical results, and angiograms. Neurosurgery 32 : 527-531, 1993
5) 宝金清博, 中川翼, 上山博康, ほか : もやもや病に対する血行再建術. 脳神経外科27 : 211-222, 1999
3) Houkin K, Kamiyama H, Takahashi A, et al : Combined revascularization surgery for childhood moyamoya disease : STA-MCA and encephalo-duro-arterio-myo-synangiosis. Childs Nery Syst 13 : 24-29, 1997
12) Kuroda S, Kamiyama H, Abe H, et al : Cerebral blood flow in children with spontaneous occlusion of the circle of Willis (moyamoya disease) : comparison with healthy children and evaluation of annual changes. Neurol Med Chir (Tokyo) 33 : 434-438, 1993
References_xml – reference: 22) Touho H, Karasawa J, Ohnishi H : Haemodynamic evaluation of parapretic transient ischaemic attacks in childhood movamova disease. Neurol Res 17 : 162-168, 1995
– reference: 11) Kuroda S, Kamiyama H, Abe H, et al : Acetazolamide test in detecting reduced cerebral perfusion reserve and predicting long-term prognosis in patients with internal carotid artery occlusion. Neurosurgery 32 : 912-919, 1993
– reference: 21) Takahashi A, Kamiyama H, Houkin K et al : Surgical treatment of childhood moyamoya disease-comparison of reconstructive surgery centered on the frontal region and the parietal region-. Neurol Med Chir (Tokyo) 35 : 231-237, 1995
– reference: 4) 宝金清博, 石川達哉, 高橋明弘, ほか : 小児もやもや病外科治療上の問題点-残された問題は何か?-. 脳卒中の外科26 : 38-44, 1998
– reference: 5) 宝金清博, 中川翼, 上山博康, ほか : もやもや病に対する血行再建術. 脳神経外科27 : 211-222, 1999
– reference: 17) Matsushima Y, Aoyagi M, Nariai T, et al : Long-term intelligence outcome of post-encephalo-duro-arterio-synangiosis childhood moyamoya disease. Clin Neurol Neurosurg 99 (Suppl 2) : S147-S150, 1997
– reference: 12) Kuroda S, Kamiyama H, Abe H, et al : Cerebral blood flow in children with spontaneous occlusion of the circle of Willis (moyamoya disease) : comparison with healthy children and evaluation of annual changes. Neurol Med Chir (Tokyo) 33 : 434-438, 1993
– reference: 8) Karasawa J, Touho H, Ohnishi H, et al : Long-term followup study after extracranial-intracranial bypass surgery for anterior circulation ischemia in childhood moyamoya disease. J Neurosurg 77 : 84-89, 1992
– reference: 10) Kinugasa K, Mandai S, Kamata I, et al : Surgical treatment of moyamoya disease : operative technique for encephaloduro-arterio-myo-synangiosis, its follow-up, clinical results, and angiograms. Neurosurgery 32 : 527-531, 1993
– reference: 20) Suzuki J, Takaku A : Cerebrovascular “moyamoya” disease : disease showing abnormal net-like vessels in the base of brain. Arch Neurol 20 : 288-299, 1969
– reference: 23) 吉井与志彦 : Moya-moyaの予後に関する研究 (I)-臨床予後について-. 脳神経29 : 421-424, 1977
– reference: 2) Fukuyama Y, Umezu R : Clinical and cerebral angiographic evaluations of idiopathic progressive occlusive disease of the circle of Willis ('moyamoya'disease) in children. Brain Dev 7 : 21-37, 1985
– reference: 16) Matsushima T, Inoue TK, Suzuki SO et al : Surgical techniques and the results of a fronto-temporo-parietal combined indirect bypass procedure for children with moyamoya disease : a comparison with the results of encephalo-duro-arterio-synangiosis alone. Clin Neurol Neuro-surg 99 (Suppl 2) : S123-S127, 1997
– reference: 18) Nakashima H, Meguro T, Kawada S, et al : Long-term results of surgically treated moyamoya disease. Clin Neurol Neurosurg 99 (Suppl 2) : S156-S161, 1997
– reference: 6) Imaizumi T, Hayashi K, Saito K, et al : Long-term outcomes of pediatric moyamoya disease monitored to adulthood. Pediatr Neurol 18 : 321-325, 1998
– reference: 14) Kuroda S, Houkin K, Kamiyama H, et al : Regional cerebral hemodynamics in childhood moyamoya disease. Childs Nery Syst 11 : 584-590, 1995
– reference: 7) Ishikawa T, Houkin K, Kamiyama H, et al : Effects of surgical revascularization on outcome of patients with pediatric moyamoya disease. Stroke 28 : 1170-1173, 1997
– reference: 15) Kurokawa T, Tomita S, Ueda K, et al : Prognosis of occlusive disease of the circle of Willis (moyamoya disease) in children. Pediatr Neurol 1 : 274-277, 1985
– reference: 3) Houkin K, Kamiyama H, Takahashi A, et al : Combined revascularization surgery for childhood moyamoya disease : STA-MCA and encephalo-duro-arterio-myo-synangiosis. Childs Nery Syst 13 : 24-29, 1997
– reference: 13) Kuroda S, Kamiyama H, Houkin K, et al : Cerebral hemodynamics and“re-build up”phenomenon on electroen-cephalogram in children with moyamoya disease. Childs Nery Syst 11 : 214-219, 1995
– reference: 1) Fukui M : Current state of study on moyamoya disease in Japan. Surg Neurol 47 : 138-143, 1997
– reference: 9) Komiyama M, Yasui T, Kitano S, et al : Moyamoya disease and pregnancy : case report and review of the literature. Neurosurgery 43 : 360-369, 1998
– reference: 19) Sato H, Sato N, Tamaki N, et al : Chronic low-perfusion state in children with moyamoya disease following revascularization. Childs Nery Syst 6 : 166-171, 1990
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Snippet もやもや病(ウイリス動脈輪閉塞症)は疾患概念の確立から約30年が経過し20), その病因, 病態, 治療法などが年々明らかとなりつつある1). 本疾患は小児にも好発し脳虚血発...
We assessed long-term outcome after surgical revascularization in childhood moyamoya disease. During the past 20 years, 51 children with moyamoya disease were...
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StartPage 421
SubjectTerms cerebral ischemia
long-term outcome
moyamoya disease
STA-MCA anastomosis
surgical revascularization
Subtitle 当科における20年間の経験から
Title 小児もやもや病の術後長期成績
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