限局性皮質異形成を発作起源とする難治性てんかん重積を呈したキャッスルマン病の1例

症例は数年前より血中IL-6値の異常高値が確認されていた55歳男性である．痙攣発作で入院し，血中および髄液IL-6値の異常高値と，FDG-PETで左前頭葉皮質の一部に集積亢進像が確認された．同部位はしだいに脳波異常を呈し，脳生検で限局性皮質異形成と診断されたが，てんかん治療にもかかわらず，頭部MRIT2強調画像で高信号を呈して拡大した．その後，腹部リンパ節生検でキャッスルマン病と確定診断されたが，第58病日に死亡した．キャッスルマン病による慢性的なIL-6過剰分泌が，限局性皮質異形成による難治性てんかんの背景因子となった可能性が考えられた．...

Full description

Saved in:
Bibliographic Details
Published in臨床神経学 Vol. 51; no. 7; pp. 487 - 492
Main Authors 松井, 真, 立花, 修, 木下, 英理子, 垣内, 無一, 岩男, 悠, 舟木, 洋
Format Journal Article
LanguageJapanese
Published 日本神経学会 2011
Subjects
IL-6
てんかん原性
てんかん重積
キャッスルマン病
限局性皮質異形成
Online AccessGet full text
ISSN0009-918X
1882-0654
DOI10.5692/clinicalneurol.51.487

Cover

Abstract 症例は数年前より血中IL-6値の異常高値が確認されていた55歳男性である．痙攣発作で入院し，血中および髄液IL-6値の異常高値と，FDG-PETで左前頭葉皮質の一部に集積亢進像が確認された．同部位はしだいに脳波異常を呈し，脳生検で限局性皮質異形成と診断されたが，てんかん治療にもかかわらず，頭部MRIT2強調画像で高信号を呈して拡大した．その後，腹部リンパ節生検でキャッスルマン病と確定診断されたが，第58病日に死亡した．キャッスルマン病による慢性的なIL-6過剰分泌が，限局性皮質異形成による難治性てんかんの背景因子となった可能性が考えられた．
AbstractList 症例は数年前より血中IL-6値の異常高値が確認されていた55歳男性である．痙攣発作で入院し，血中および髄液IL-6値の異常高値と，FDG-PETで左前頭葉皮質の一部に集積亢進像が確認された．同部位はしだいに脳波異常を呈し，脳生検で限局性皮質異形成と診断されたが，てんかん治療にもかかわらず，頭部MRIT2強調画像で高信号を呈して拡大した．その後，腹部リンパ節生検でキャッスルマン病と確定診断されたが，第58病日に死亡した．キャッスルマン病による慢性的なIL-6過剰分泌が，限局性皮質異形成による難治性てんかんの背景因子となった可能性が考えられた．
Author 松井, 真
垣内, 無一
立花, 修
木下, 英理子
岩男, 悠
舟木, 洋
Author_xml – sequence: 1
  fullname: 松井, 真
  organization: 金沢医科大学神経内科
– sequence: 1
  fullname: 立花, 修
  organization: 金沢医科大学脳神経外科
– sequence: 1
  fullname: 木下, 英理子
  organization: 金沢医科大学臨床病理
– sequence: 1
  fullname: 垣内, 無一
  organization: 金沢医科大学神経内科
– sequence: 1
  fullname: 岩男, 悠
  organization: 金沢医科大学血液免疫内科
– sequence: 1
  fullname: 舟木, 洋
  organization: 金沢医科大学消化器外科
BookMark eNpVkN1KAlEUhQ9RkJmP0COMnfk_cxnSL0I3Bd0Nx8OxRiaN0YLunBkiQQQrQqwoSOiXpJKIDHuZrY6-RRMG0c1abNa3N-w1hcazuSxHaEbEcVUzpFlmW1mLUTvLd52cHVfFuEL0MRQRCZEErKnKOIpgjA3BEMnGJIrl81bqZ1YNIioRtDesV3svxX7xNjhrDloPwelzr3PdL1XBOw7q7W7nYvD23m9Xwb0Dtw5eeXje6L9-hjy4N-CdgFsOdXhYCe4r4UrvqARuDdwr8J7Ab4Dvg_cB_iP4l-C3gtoBuE2x-1WeRhNpaud57NejaH1hfi2xJCRXF5cTc0khI6kEC5wzQ5eJomFJxYpuUMYY1hiRdUXR5FQaUzH8hcq6niYG05jBMQ1zpujpVIrLkhxFK6O7mXyBbnJzx7G2qbNvUqdgMZub_9szVdHURxKW-AdtUcfMUPkbWHSasA
ContentType Journal Article
Copyright 2011 日本神経学会
Copyright_xml – notice: 2011 日本神経学会
DOI 10.5692/clinicalneurol.51.487
DatabaseTitleList
DeliveryMethod fulltext_linktorsrc
Discipline Medicine
EISSN 1882-0654
EndPage 492
ExternalDocumentID article_clinicalneurol_51_7_51_7_487_article_char_ja
GroupedDBID ALMA_UNASSIGNED_HOLDINGS
JSF
OK1
P2P
RJT
ID FETCH-LOGICAL-j2580-eec9738460250479accc06c8374463bf0a1000a377f89c6c9e0a06cc47fbbe323
ISSN 0009-918X
IngestDate Wed Sep 03 06:19:35 EDT 2025
IsDoiOpenAccess true
IsOpenAccess true
IsPeerReviewed true
IsScholarly true
Issue 7
Language Japanese
LinkModel OpenURL
MergedId FETCHMERGED-LOGICAL-j2580-eec9738460250479accc06c8374463bf0a1000a377f89c6c9e0a06cc47fbbe323
OpenAccessLink https://www.jstage.jst.go.jp/article/clinicalneurol/51/7/51_7_487/_article/-char/ja
PageCount 6
ParticipantIDs jstage_primary_article_clinicalneurol_51_7_51_7_487_article_char_ja
PublicationCentury 2000
PublicationDate 20110000
PublicationDateYYYYMMDD 2011-01-01
PublicationDate_xml – year: 2011
  text: 20110000
PublicationDecade 2010
PublicationTitle 臨床神経学
PublicationTitleAlternate 臨床神経学
PublicationYear 2011
Publisher 日本神経学会
Publisher_xml – name: 日本神経学会
References 2) Palmini A, Najm I, Avanzini G, et al. Terminology and classification of the cortical dysplasias. Neurology 2004;62:S2-8.
7) 柳下 章, 新井信隆. 難治性てんかんの画像と病理. 第1版. 東京: 秀潤社; 2007. p. 95-97.
8) Yagishita A, Arai N, Maehara T, et al. Focal cortical dysplasia: appearance on MR images. Radiology 1997;203:553-559.
16) Severson GS, Harrington DS, Weisenburger DD, et al. Castleman's disease of the leptomeninges. Report of three cases. J Neurosurg 1988;69:283-286.
20) Rodgers KM, Hutchinson MR, Northcutt A, et al. The cortical innate immune response increases local neuronal excitability leading to seizures. Brain 2009;132:2478-2486.
6) Peterson BA, Frizzera G. Multicentric Castleman's disease. Semin Oncol 1993;20:636-647.
19) Vezzani A, Granata T. Brain inflammation in epilepsy: experimental and clinical evidence. Epilepsia 2005;46:1724-1743.
18) Mallik AA, Katchy KC, Clotan N. Solitary intracranial Castleman's disease, plasma cell variant: a case report. Med Princ Pract 2007;16:226-229.
4) 松田一己, 久保田裕子. 腫瘍様の形成異常とてんかん原性病変. 病理と臨床 2004;22:1010-1016.
9) Cronin DM, Warnke RA. Castleman disease: an update on classification and the spectrum of associated lesions. Adv Anat Pathol 2009;16:236-246.
12) Coca S, Salas I, Martínez R, et al. Meningeal Castleman's disease with multifocal involvement: a case report and review of literature. J Neurooncol 2008;88:37-41.
11) Nishimoto N, Kanakura Y, Aozasa K, et al. Humanized anti-interleukin-6 receptor antibody treatment of multicentric Castleman disease. Blood 2005;106:2627-2632.
3) Lawson JA, Birchansky S, Pacheco E, et al. Distinct clinicopathologic subtypes of cortical dysplasia of Taylor. Neurology 2005;64:55-61.
10) 中塚伸一, 堀由美子, 青笹克之. キャッスルマン病の病理診断と鑑別. Pharma Medica 2009;27:93-100.
15) Matsumura K, Nakasu S, Tanaka T, et al. Intracranial localized Castleman's disease. Case report. Neurol Med Chir (Tokyo) 2005;45:59-65.
1) Taylor DC, Falconer MA, Bruton CJ, et al. Focal cortical dysplasia of the cerebral cortex in epilepsy. J Neurol Neurosurg Psychiatry 1971;34:369-387.
14) Black DA, Forgacs I, Davies DR, et al. Pseudotumour cerebri in a patient with Castleman's disease. Postgrad Med J 1988;64:217-219.
21) Vezzani A, Balosso S, Ravizza T. The role of cytokines in the pathophysiology of epilepsy. Brain Behav Immun 2008;22:797-803.
5) Castleman B, Towne V. Case records of the Massachusetts General Hospital: Case No. 40231. N Engl J Med 1954;250:1001-1005.
13) Shuaipaj T, Abutalib SA, Chen YH, et al. Intracranial plasmacytoma mimicking meningioma in a patient with Castleman's disease. Am J Hematol 2009;84:195-196.
17) Cummings TJ, Gong JZ, Friedman AH, et al. Castleman's disease confined to the leptomeninges. Ann Clin Lab Sci 2000;30:278-282.
References_xml – reference: 18) Mallik AA, Katchy KC, Clotan N. Solitary intracranial Castleman's disease, plasma cell variant: a case report. Med Princ Pract 2007;16:226-229.
– reference: 7) 柳下 章, 新井信隆. 難治性てんかんの画像と病理. 第1版. 東京: 秀潤社; 2007. p. 95-97.
– reference: 3) Lawson JA, Birchansky S, Pacheco E, et al. Distinct clinicopathologic subtypes of cortical dysplasia of Taylor. Neurology 2005;64:55-61.
– reference: 17) Cummings TJ, Gong JZ, Friedman AH, et al. Castleman's disease confined to the leptomeninges. Ann Clin Lab Sci 2000;30:278-282.
– reference: 20) Rodgers KM, Hutchinson MR, Northcutt A, et al. The cortical innate immune response increases local neuronal excitability leading to seizures. Brain 2009;132:2478-2486.
– reference: 5) Castleman B, Towne V. Case records of the Massachusetts General Hospital: Case No. 40231. N Engl J Med 1954;250:1001-1005.
– reference: 6) Peterson BA, Frizzera G. Multicentric Castleman's disease. Semin Oncol 1993;20:636-647.
– reference: 9) Cronin DM, Warnke RA. Castleman disease: an update on classification and the spectrum of associated lesions. Adv Anat Pathol 2009;16:236-246.
– reference: 14) Black DA, Forgacs I, Davies DR, et al. Pseudotumour cerebri in a patient with Castleman's disease. Postgrad Med J 1988;64:217-219.
– reference: 15) Matsumura K, Nakasu S, Tanaka T, et al. Intracranial localized Castleman's disease. Case report. Neurol Med Chir (Tokyo) 2005;45:59-65.
– reference: 21) Vezzani A, Balosso S, Ravizza T. The role of cytokines in the pathophysiology of epilepsy. Brain Behav Immun 2008;22:797-803.
– reference: 13) Shuaipaj T, Abutalib SA, Chen YH, et al. Intracranial plasmacytoma mimicking meningioma in a patient with Castleman's disease. Am J Hematol 2009;84:195-196.
– reference: 2) Palmini A, Najm I, Avanzini G, et al. Terminology and classification of the cortical dysplasias. Neurology 2004;62:S2-8.
– reference: 1) Taylor DC, Falconer MA, Bruton CJ, et al. Focal cortical dysplasia of the cerebral cortex in epilepsy. J Neurol Neurosurg Psychiatry 1971;34:369-387.
– reference: 11) Nishimoto N, Kanakura Y, Aozasa K, et al. Humanized anti-interleukin-6 receptor antibody treatment of multicentric Castleman disease. Blood 2005;106:2627-2632.
– reference: 19) Vezzani A, Granata T. Brain inflammation in epilepsy: experimental and clinical evidence. Epilepsia 2005;46:1724-1743.
– reference: 4) 松田一己, 久保田裕子. 腫瘍様の形成異常とてんかん原性病変. 病理と臨床 2004;22:1010-1016.
– reference: 8) Yagishita A, Arai N, Maehara T, et al. Focal cortical dysplasia: appearance on MR images. Radiology 1997;203:553-559.
– reference: 10) 中塚伸一, 堀由美子, 青笹克之. キャッスルマン病の病理診断と鑑別. Pharma Medica 2009;27:93-100.
– reference: 12) Coca S, Salas I, Martínez R, et al. Meningeal Castleman's disease with multifocal involvement: a case report and review of literature. J Neurooncol 2008;88:37-41.
– reference: 16) Severson GS, Harrington DS, Weisenburger DD, et al. Castleman's disease of the leptomeninges. Report of three cases. J Neurosurg 1988;69:283-286.
SSID ssib000959814
ssib058494234
ssib002821941
ssib000940416
ssib002484599
ssib005879791
ssj0060813
Score 1.8680888
Snippet ...
SourceID jstage
SourceType Publisher
StartPage 487
SubjectTerms IL-6
てんかん原性
てんかん重積
キャッスルマン病
限局性皮質異形成
Title 限局性皮質異形成を発作起源とする難治性てんかん重積を呈したキャッスルマン病の1例
URI https://www.jstage.jst.go.jp/article/clinicalneurol/51/7/51_7_487/_article/-char/ja
Volume 51
hasFullText 1
inHoldings 1
isFullTextHit
isPrint
ispartofPNX 臨床神経学, 2011, Vol.51(7), pp.487-492
link http://utb.summon.serialssolutions.com/2.0.0/link/0/eLvHCXMwrV1baxQxFB5KBfFFvOKdPpjHrXPLJHkzs51SFAWhhb4ts9NdZJEqpfrgU3cXsVAKVZFSFQULXrF4QcRK_TNpd9t_4TmZzOysFNEiLCEk3zk552R2Tk4mF8s679dZ7CY0LlXBeZf8JKAlzgXTG3WFV3fwsgdcbXE1GJvwL03SyYHBi8XdJbPV4eTurvtK9tKrUAb9irtk_6Fnc6ZQAHnoX0ihhyH9qz4mkSACfjaJKAkdXLUQBZhKRiJGhCQyIhEnoUek1CUAS8EjRLoazDW5R7hLhKsxgoQA9hEjypoc8AzBUG7ADpHcZACfkvNQyxMS6WmwS8KwIE9KFWRteaYEqfISQTgjfATFkEJncsEoEQ5KaxrNGIpRg5Ep2NOt6wz3TFUosqrQZERkMmAZVBkapZmEkaOVj0Cy4sgdDQHCodoUDcG1QSXVvBjaiJexCuSQ-Wwnqi9GkBmylGh_eKR1k2UQvQdjKBuagiNj6EoNA5pREKiPW1mLADUc8Rqmc0CDbG3CAyOGsHuEWky0DMV6novBfSKdjJ1dnAJyepM_ul2tqxGg_AfVNTNQTvb5QwHuTl_uDKOB1AVizIVbjos-0hwKnL4LWMHh-Wa4lI6d_PRew9_dMg0EHvOb7fXVJ7XeGKbOcE7dd-K5-T9V-vEV6lRYmgBZJQddj2cqDYiQ9rmMObio9_K1QoAhfLvvu7qgghfWDbg-96nom4Bwiju7KWeC9U6TgtG6gPjDz8Z6AbzJ8rsa0Y7pHkHU98Ku2sI4twFRX7ZiVA9ixw9ZB030OSRTrQ5bA434iLX_illfc9S6s7OytPVprjP3uvtkbfvLu-7jj1sbLzvzS6r1sLuyvrnxbPvrt876kmq-Uc0V1VrYebra-fwD8Kr5SrUeqeYCpDv3F7tvF4Fk68G8ai6r5gvV-qDaq6rdVq3vqv1etZ-r9pfu8j3VXHM2fy4csyZGo_HyWMnczFJquJTbpVotEcyD0EWfgMhEnCSJHSTcY74feNW6HeNnw9hjrM5FEiSiZsdQn_isXq3WPNc7bg1O35yunbCG6lWvNmVzeN7qiR84QoBLgSAkSFxhT3lT4qRVTi1WuZUev1PZy-Nx6r9wOW0dSD8p4e-MNTg7c7t2FmKS2eo5_dj9AuR8DGI
linkProvider Colorado Alliance of Research Libraries
openUrl ctx_ver=Z39.88-2004&ctx_enc=info%3Aofi%2Fenc%3AUTF-8&rfr_id=info%3Asid%2Fsummon.serialssolutions.com&rft_val_fmt=info%3Aofi%2Ffmt%3Akev%3Amtx%3Ajournal&rft.genre=article&rft.atitle=%E9%99%90%E5%B1%80%E6%80%A7%E7%9A%AE%E8%B3%AA%E7%95%B0%E5%BD%A2%E6%88%90%E3%82%92%E7%99%BA%E4%BD%9C%E8%B5%B7%E6%BA%90%E3%81%A8%E3%81%99%E3%82%8B%E9%9B%A3%E6%B2%BB%E6%80%A7%E3%81%A6%E3%82%93%E3%81%8B%E3%82%93%E9%87%8D%E7%A9%8D%E3%82%92%E5%91%88%E3%81%97%E3%81%9F%E3%82%AD%E3%83%A3%E3%83%83%E3%82%B9%E3%83%AB%E3%83%9E%E3%83%B3%E7%97%85%E3%81%AE1%E4%BE%8B&rft.jtitle=%E8%87%A8%E5%BA%8A%E7%A5%9E%E7%B5%8C%E5%AD%A6&rft.au=%E6%9D%BE%E4%BA%95%2C+%E7%9C%9F&rft.au=%E7%AB%8B%E8%8A%B1%2C+%E4%BF%AE&rft.au=%E6%9C%A8%E4%B8%8B%2C+%E8%8B%B1%E7%90%86%E5%AD%90&rft.au=%E5%9E%A3%E5%86%85%2C+%E7%84%A1%E4%B8%80&rft.date=2011&rft.pub=%E6%97%A5%E6%9C%AC%E7%A5%9E%E7%B5%8C%E5%AD%A6%E4%BC%9A&rft.issn=0009-918X&rft.eissn=1882-0654&rft.volume=51&rft.issue=7&rft.spage=487&rft.epage=492&rft_id=info:doi/10.5692%2Fclinicalneurol.51.487&rft.externalDocID=article_clinicalneurol_51_7_51_7_487_article_char_ja
thumbnail_l http://covers-cdn.summon.serialssolutions.com/index.aspx?isbn=/lc.gif&issn=0009-918X&client=summon
thumbnail_m http://covers-cdn.summon.serialssolutions.com/index.aspx?isbn=/mc.gif&issn=0009-918X&client=summon
thumbnail_s http://covers-cdn.summon.serialssolutions.com/index.aspx?isbn=/sc.gif&issn=0009-918X&client=summon