5年以上経過を追えた自己免疫性自律神経節障害の3例
5年以上経過を追えた抗自律神経節アセチルコリン受容体抗体陽性で自己免疫性自律神経節障害（autoimmune autonomic ganglionopathy，以下AAGと略記）と診断された3例の経過を報告する．1例目は20代女性で慢性経過の羞明・便秘・無月経を認めたが，血漿交換後，症状は緩解した．感冒を契機に一度再発したが，2度の妊娠による悪化はなかった．2例目は60代男性で急性の起立性低血圧（orthostatic hypotension，以下OHと略記）と精神症状で発症した．再燃を繰り返し治療に難渋したが，経過8年時，特に原因なく状態が安定した．3例目は80代女性で慢性経過のOHで再燃を...
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| Published in | 臨床神経学 Vol. 62; no. 11; pp. 860 - 864 |
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| Main Authors | , , , , |
| Format | Journal Article |
| Language | Japanese |
| Published |
日本神経学会
2022
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| Subjects | |
| Online Access | Get full text |
| ISSN | 0009-918X 1882-0654 |
| DOI | 10.5692/clinicalneurol.cn-001793 |
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| Abstract | 5年以上経過を追えた抗自律神経節アセチルコリン受容体抗体陽性で自己免疫性自律神経節障害（autoimmune autonomic ganglionopathy，以下AAGと略記）と診断された3例の経過を報告する．1例目は20代女性で慢性経過の羞明・便秘・無月経を認めたが，血漿交換後，症状は緩解した．感冒を契機に一度再発したが，2度の妊娠による悪化はなかった．2例目は60代男性で急性の起立性低血圧（orthostatic hypotension，以下OHと略記）と精神症状で発症した．再燃を繰り返し治療に難渋したが，経過8年時，特に原因なく状態が安定した．3例目は80代女性で慢性経過のOHで再燃を繰り返した．経過中，OHによる転倒で大腿骨を骨折し歩行困難となった．AAGの長期観察例の報告は少なく，臨床経過を観察する上で貴重と考え報告する． |
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| AbstractList | 5年以上経過を追えた抗自律神経節アセチルコリン受容体抗体陽性で自己免疫性自律神経節障害（autoimmune autonomic ganglionopathy，以下AAGと略記）と診断された3例の経過を報告する．1例目は20代女性で慢性経過の羞明・便秘・無月経を認めたが，血漿交換後，症状は緩解した．感冒を契機に一度再発したが，2度の妊娠による悪化はなかった．2例目は60代男性で急性の起立性低血圧（orthostatic hypotension，以下OHと略記）と精神症状で発症した．再燃を繰り返し治療に難渋したが，経過8年時，特に原因なく状態が安定した．3例目は80代女性で慢性経過のOHで再燃を繰り返した．経過中，OHによる転倒で大腿骨を骨折し歩行困難となった．AAGの長期観察例の報告は少なく，臨床経過を観察する上で貴重と考え報告する． |
| Author | 鳥飼, 裕子 原, 一 黒野, 裕子 岡村, 正哉 國本, 雅也 |
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| References | 4) Yamakawa M, Watari M, Torii K, et al. gAChR antibodies in children and adolescents with acquired autoimmune dysautonomia in Japan. Ann Clin Transl Neurol 2021;8:790-799. 3) Nakane S, Mukaino A, Higuchi O, et al. A comprehensive analysis of the clinical characteristics and laboratory features in 179 patients with autoimmune autonomic ganglionopathy. J Autoimmun 2020;108;102403. 1) Vernino S, Low PA, Fealey RD, et al. Autoantibodies to ganglionic acetylcholine receptors in autoimmune autonomic neuropathies. N Engl J Med 2000;343:847-855. 12) Mobeen H, Afzal N, Kashif M. Polycystic ovary syndrome may be an autoimmune disorder. Scientifica (Cairo). 2016;2016:4071735. 13) Romitti M, Fabris VC, Ziegelmann PK, et al. Association between PCOS and autoimmune thyroid disease: a systematic review and meta-analysis. Endocr Connect 2018;7:1158-1167. 8) Li Y, Jammoul A, Mente K, et al. Clinical experience of seropositive ganglionic acetylcholine receptor antibody in a tertiary neurology referral center. Muscle Nerve 2015;52:386-391. 10) Schroeder C, Vernino S, Birkenfeld AL, et al. Plasma exchange for primary autoimmune autonomic failure. N Engl J Med 2005;353:1585-1590. 5) 山川誠，中根俊成．自己免疫性自律神経節障害：10の課題．末梢神経 2021;32:38-46. 7) Vernino S, Low PA, Lennon VA. Experimental autoimmune autonomic neuropathy. J Neurophysiol 2003;90:2053-2059. 6) Tajzoy E, Mukherjee S, Vernino S. Autonomic ganglia neuronal density and synaptic structure in chronic experimental autoimmune autonomic ganglionopathy. Arch Neurol 2011;68:540-541. 9) Gibbons CH, Freeman R. Antibody titers predict clinical features of autoimmune autonomic ganglionopathy. Auton Neurosci 2009;146:8-12. 11) 中根俊成．自己免疫性自律神経節障害．autoimmune autonomic ganglionopathy．臨床神経 2013;53:1071-1073. 14) 中根俊成，安東由喜雄．アセチルコリン受容体に対する自己抗体と脳炎・脳症．日内会誌 2017;106:1571-1578. 2) Nakane S, Higuchi O, Koga M, et al. Clinical features of autoimmune autonomic ganglionopathy and the detection of subunit-specific autoantibodies to the ganglionic acetylcholine receptor in Japanese patients. PLoS One 2015;10:e0118312. |
| References_xml | – reference: 12) Mobeen H, Afzal N, Kashif M. Polycystic ovary syndrome may be an autoimmune disorder. Scientifica (Cairo). 2016;2016:4071735. – reference: 1) Vernino S, Low PA, Fealey RD, et al. Autoantibodies to ganglionic acetylcholine receptors in autoimmune autonomic neuropathies. N Engl J Med 2000;343:847-855. – reference: 7) Vernino S, Low PA, Lennon VA. Experimental autoimmune autonomic neuropathy. J Neurophysiol 2003;90:2053-2059. – reference: 8) Li Y, Jammoul A, Mente K, et al. Clinical experience of seropositive ganglionic acetylcholine receptor antibody in a tertiary neurology referral center. Muscle Nerve 2015;52:386-391. – reference: 10) Schroeder C, Vernino S, Birkenfeld AL, et al. Plasma exchange for primary autoimmune autonomic failure. N Engl J Med 2005;353:1585-1590. – reference: 11) 中根俊成．自己免疫性自律神経節障害．autoimmune autonomic ganglionopathy．臨床神経 2013;53:1071-1073. – reference: 2) Nakane S, Higuchi O, Koga M, et al. Clinical features of autoimmune autonomic ganglionopathy and the detection of subunit-specific autoantibodies to the ganglionic acetylcholine receptor in Japanese patients. PLoS One 2015;10:e0118312. – reference: 13) Romitti M, Fabris VC, Ziegelmann PK, et al. Association between PCOS and autoimmune thyroid disease: a systematic review and meta-analysis. Endocr Connect 2018;7:1158-1167. – reference: 14) 中根俊成，安東由喜雄．アセチルコリン受容体に対する自己抗体と脳炎・脳症．日内会誌 2017;106:1571-1578. – reference: 4) Yamakawa M, Watari M, Torii K, et al. gAChR antibodies in children and adolescents with acquired autoimmune dysautonomia in Japan. Ann Clin Transl Neurol 2021;8:790-799. – reference: 9) Gibbons CH, Freeman R. Antibody titers predict clinical features of autoimmune autonomic ganglionopathy. Auton Neurosci 2009;146:8-12. – reference: 3) Nakane S, Mukaino A, Higuchi O, et al. A comprehensive analysis of the clinical characteristics and laboratory features in 179 patients with autoimmune autonomic ganglionopathy. J Autoimmun 2020;108;102403. – reference: 5) 山川誠，中根俊成．自己免疫性自律神経節障害：10の課題．末梢神経 2021;32:38-46. – reference: 6) Tajzoy E, Mukherjee S, Vernino S. Autonomic ganglia neuronal density and synaptic structure in chronic experimental autoimmune autonomic ganglionopathy. Arch Neurol 2011;68:540-541. |
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| SubjectTerms | 自己免疫性自律神経節障害 自律神経障害 長期経過観察 |
| Title | 5年以上経過を追えた自己免疫性自律神経節障害の3例 |
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