Type B3胸腺腫を合併した抗gamma aminobutyric acid （GABA）A受容体抗体陽性再発性脳炎の1例

症例は87歳女性である．亜急性に進行する認知機能低下および発語量低下を呈し当科を初診となった．来院時の頭部造影MRIではT2強調画像にて多発する高信号病変を大脳皮質および白質に認めた．胸部CT検査では胸腺腫を指摘され，血清中抗gamma aminobutyric acid (GABA)A受容体抗体陽性であったことから，胸腺腫合併抗GABAA受容体抗体陽性脳炎と診断した．胸腺腫摘出および血漿交換/ステロイド療法により症状は完全寛解を示したが，ステロイド漸減に伴い脳炎の再発を繰り返した．抗GABAA受容体抗体陽性脳炎の報告は世界的にも極めて稀であり，治療経過とともに報告する．...

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Published in臨床神経学 Vol. 56; no. 11; pp. 764 - 768
Main Authors 伏見, 博彰, 木下, 允, 北野, 貴也, 大森, 謙一, 島津, 宏樹, 狭間, 敬憲
Format Journal Article
LanguageJapanese
Published 日本神経学会 2016
Subjects
抗gamma aminobutyric acid (GABA)A受容体抗体
胸腺腫
脳炎
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ISSN0009-918X
1882-0654
DOI10.5692/clinicalneurol.cn-000930

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Abstract 症例は87歳女性である．亜急性に進行する認知機能低下および発語量低下を呈し当科を初診となった．来院時の頭部造影MRIではT2強調画像にて多発する高信号病変を大脳皮質および白質に認めた．胸部CT検査では胸腺腫を指摘され，血清中抗gamma aminobutyric acid (GABA)A受容体抗体陽性であったことから，胸腺腫合併抗GABAA受容体抗体陽性脳炎と診断した．胸腺腫摘出および血漿交換/ステロイド療法により症状は完全寛解を示したが，ステロイド漸減に伴い脳炎の再発を繰り返した．抗GABAA受容体抗体陽性脳炎の報告は世界的にも極めて稀であり，治療経過とともに報告する．
AbstractList 症例は87歳女性である．亜急性に進行する認知機能低下および発語量低下を呈し当科を初診となった．来院時の頭部造影MRIではT2強調画像にて多発する高信号病変を大脳皮質および白質に認めた．胸部CT検査では胸腺腫を指摘され，血清中抗gamma aminobutyric acid (GABA)A受容体抗体陽性であったことから，胸腺腫合併抗GABAA受容体抗体陽性脳炎と診断した．胸腺腫摘出および血漿交換/ステロイド療法により症状は完全寛解を示したが，ステロイド漸減に伴い脳炎の再発を繰り返した．抗GABAA受容体抗体陽性脳炎の報告は世界的にも極めて稀であり，治療経過とともに報告する．
Author 北野, 貴也
島津, 宏樹
木下, 允
大森, 謙一
狭間, 敬憲
伏見, 博彰
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  fullname: 北野, 貴也
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  fullname: 大森, 謙一
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  organization: 大阪府立急性期・総合医療センター神経内科
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References 10) Gastaldi M, Thouin A, Vincent A. Antibody-Mediated autoimmune encephalopathies and immunotherapies. Neurotherapeutics 2016;13:147-162.
3) Petit-Pedrol M, Armangue T, Peng X, et al. Encephalitis with refractory seizures, status epilepticus, and antibodies to the GABAA receptor: a case series, characterisation of the antigen, and analysis of the effects of antibodies. Lancet Neurol 2014;13:276-286.
4) Ohkawa T, Satake S, Yokoi N, et al. Identification and characterization of GABA(A) receptor autoantibodies in autoimmune encephalitis. J Neurosci 2014;34:8151-8163.
5) Simabukuro MM, Petit-Pedrol M, Castro LH, et al. GABAA receptor and LGI1 antibody encephalitis in a patient with thymoma. Neurol Neuroimmunol Neuroinflamm 2015;2:e73.
9) Fujii Y. Thymus, thymoma and myasthenia gravis. Surg Today 2013;43:461-466.
1) Ohshita T, Kawakami H, Maruyama H, et al. Voltage-gated potassium channel antibodies associated limbic encephalitis in a patient with invasive thymoma. J Neurol Sci 2006;250:167-169.
8) Okumura M, Miyoshi S, Fujii Y, et al. Clinical and functional significance of WHO classification on human thymic epithelial neoplasms: a study of 146 consecutive tumors. Am J Surg Pathol 2001;25:103-110.
2) Miyazaki Y, Hirayama M, Watanabe H, et al. Paraneoplastic encephalitis associated with myasthenia gravis and malignant thymoma. J Clin Neurosci 2012;19:336-338.
6) Graus F, Titulaer MJ, Balu R, et al. A clinical approach to diagnosis of autoimmune encephalitis. Lancet Neurol 2016;15:391-404.
7) Okumura M, Fujii Y, Shiono H, et al. Immunological function of thymoma and pathogenesis of paraneoplastic myasthenia gravis. Gen Thorac Cardiovasc Surg 2008;56:143-150.
References_xml – reference: 3) Petit-Pedrol M, Armangue T, Peng X, et al. Encephalitis with refractory seizures, status epilepticus, and antibodies to the GABAA receptor: a case series, characterisation of the antigen, and analysis of the effects of antibodies. Lancet Neurol 2014;13:276-286.
– reference: 1) Ohshita T, Kawakami H, Maruyama H, et al. Voltage-gated potassium channel antibodies associated limbic encephalitis in a patient with invasive thymoma. J Neurol Sci 2006;250:167-169.
– reference: 4) Ohkawa T, Satake S, Yokoi N, et al. Identification and characterization of GABA(A) receptor autoantibodies in autoimmune encephalitis. J Neurosci 2014;34:8151-8163.
– reference: 10) Gastaldi M, Thouin A, Vincent A. Antibody-Mediated autoimmune encephalopathies and immunotherapies. Neurotherapeutics 2016;13:147-162.
– reference: 5) Simabukuro MM, Petit-Pedrol M, Castro LH, et al. GABAA receptor and LGI1 antibody encephalitis in a patient with thymoma. Neurol Neuroimmunol Neuroinflamm 2015;2:e73.
– reference: 6) Graus F, Titulaer MJ, Balu R, et al. A clinical approach to diagnosis of autoimmune encephalitis. Lancet Neurol 2016;15:391-404.
– reference: 9) Fujii Y. Thymus, thymoma and myasthenia gravis. Surg Today 2013;43:461-466.
– reference: 7) Okumura M, Fujii Y, Shiono H, et al. Immunological function of thymoma and pathogenesis of paraneoplastic myasthenia gravis. Gen Thorac Cardiovasc Surg 2008;56:143-150.
– reference: 8) Okumura M, Miyoshi S, Fujii Y, et al. Clinical and functional significance of WHO classification on human thymic epithelial neoplasms: a study of 146 consecutive tumors. Am J Surg Pathol 2001;25:103-110.
– reference: 2) Miyazaki Y, Hirayama M, Watanabe H, et al. Paraneoplastic encephalitis associated with myasthenia gravis and malignant thymoma. J Clin Neurosci 2012;19:336-338.
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Snippet 症例は87歳女性である．亜急性に進行する認知機能低下および発語量低下を呈し当科を初診となった．来院時の頭部造影MRIではT2強調画像にて多発する高信号病変を大脳皮...
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SubjectTerms 抗gamma aminobutyric acid (GABA)A受容体抗体
胸腺腫
脳炎
Title Type B3胸腺腫を合併した抗gamma aminobutyric acid （GABA）A受容体抗体陽性再発性脳炎の1例
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