子宮内膜原発大細胞神経内分泌癌に関連する抗Hu抗体陽性の傍腫瘍性sensory neuronopathyの1例

症例は81歳女性．入院7か月前に子宮内膜原発大細胞神経内分泌癌（large cell neuroendocrine carcinoma; LCNEC）のため子宮全摘術および両側付属器切除術が施行された．入院3か月前より亜急性に四肢末梢優位の異常感覚と歩行困難が出現し当科を受診した．四肢の異常感覚，下肢振動覚低下，腱反射消失，自律神経障害を呈し，神経伝導検査で感覚神経の活動電位は導出されなかった．CTで骨盤内多発リンパ節腫大を認め，術後低下していたneuron specific enolase（NSE）の再上昇と合わせてLCNECの術後再発に伴う傍腫瘍性sensory neuronopathyと...

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Published in臨床神経学 Vol. 60; no. 6; pp. 441 - 445
Main Authors 加藤, 里奈, 蕨, 雅大, 藤ヶ崎, 浩人, 青山, 尚史, 笠松, 高弘, 水谷, 真之
Format Journal Article
LanguageJapanese
Published 日本神経学会 2020
Subjects
傍腫瘍性神経症候群
子宮内膜原発大細胞神経内分泌癌
感覚性ニューロノパチー
抗Hu抗体
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ISSN0009-918X
1882-0654
DOI10.5692/clinicalneurol.60.cn-001393

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Abstract 症例は81歳女性．入院7か月前に子宮内膜原発大細胞神経内分泌癌（large cell neuroendocrine carcinoma; LCNEC）のため子宮全摘術および両側付属器切除術が施行された．入院3か月前より亜急性に四肢末梢優位の異常感覚と歩行困難が出現し当科を受診した．四肢の異常感覚，下肢振動覚低下，腱反射消失，自律神経障害を呈し，神経伝導検査で感覚神経の活動電位は導出されなかった．CTで骨盤内多発リンパ節腫大を認め，術後低下していたneuron specific enolase（NSE）の再上昇と合わせてLCNECの術後再発に伴う傍腫瘍性sensory neuronopathyと診断した．抗Hu抗体陽性の稀な1例であり報告する．
AbstractList 症例は81歳女性．入院7か月前に子宮内膜原発大細胞神経内分泌癌（large cell neuroendocrine carcinoma; LCNEC）のため子宮全摘術および両側付属器切除術が施行された．入院3か月前より亜急性に四肢末梢優位の異常感覚と歩行困難が出現し当科を受診した．四肢の異常感覚，下肢振動覚低下，腱反射消失，自律神経障害を呈し，神経伝導検査で感覚神経の活動電位は導出されなかった．CTで骨盤内多発リンパ節腫大を認め，術後低下していたneuron specific enolase（NSE）の再上昇と合わせてLCNECの術後再発に伴う傍腫瘍性sensory neuronopathyと診断した．抗Hu抗体陽性の稀な1例であり報告する．
Author 水谷, 真之
蕨, 雅大
笠松, 高弘
藤ヶ崎, 浩人
青山, 尚史
加藤, 里奈
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14) Derks JL, van Suylen RJ, Thunnissen E, et al. Chemotherapy for pulmonary large cell neuroendocrine carcinomas: does the regimen matter? Eur Respir J 2017;49:1601838.
10) Dalmau J, Graus F, Rosenblum MK, et al. Anti-Hu-associated paraneoplastic encephalomyelitis/sensory neuronopathy. A clinical study of 71 patients. Medicine 1992;71:59-72.
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8) Pignolet BS, Gebauer CM, Liblau RS. Immunopathogenesis of paraneoplastic neurological syndromes associated with anti-Hu antibodies: A beneficial antitumor immune response going awry. Oncoimmunology 2013;2:e27384.
13) Ishikawa M, Kasamatsu T, Tsuda H, et al. Prognostic factors and optimal therapy for stage I-II neuroendocrine carcinomas of the uterine cervix: a multi-center retrospective study. Gynecol Oncol 2018;148:139-146.
7) Gordon WC, Leach MTJ. Lung carcinoma with anti-Hu paraneoplastic syndrome. N Eng J Med 2006;355:e4.
11) George J, Walter V, Peifer M, et al. Integrative genomic profiling of large-cell neuroendocrine carcinomas reveals distinct subtypes of high-grade neuroendocrine lung tumors. Nat Commun 2018;9:1048-1060.
4) Ogura J, Adachi Y, Yasumoto K, et al. Large-cell neuro­endocrine carcinoma arising in the endometrium: a case report. Mol Clin Oncol 2018;8:575-578.
9) Zaborowski MP, Michalak S. Cell-mediated immune responses in paraneoplastic neurological syndromes. Clin Dev Immunol 2013;630602:11.
References_xml – reference: 8) Pignolet BS, Gebauer CM, Liblau RS. Immunopathogenesis of paraneoplastic neurological syndromes associated with anti-Hu antibodies: A beneficial antitumor immune response going awry. Oncoimmunology 2013;2:e27384.
– reference: 3) 田中恵子．傍腫瘍性神経症候群．医のあゆみ 2015;255:543-547.
– reference: 1) 田中恵子．傍腫瘍性神経症候群の診断と治療．神経治療 2014;31:153-155.
– reference: 2) Graus F, Delattre JY, Antoine JC, et al. Recommended diagnostic criteria for paraneoplastic neurological syndromes. J Neural Neurosurg Psychiatry 2004;75:1135-1140.
– reference: 5) Camdessanche JP, Antoine JC, Honnorat J, et al. Paraneoplastic peripheral neuropathy associated with anti-Hu antibodies. A clinical and electrophysiological study of 20 patients. Brain 2002;125:166-175.
– reference: 10) Dalmau J, Graus F, Rosenblum MK, et al. Anti-Hu-associated paraneoplastic encephalomyelitis/sensory neuronopathy. A clinical study of 71 patients. Medicine 1992;71:59-72.
– reference: 7) Gordon WC, Leach MTJ. Lung carcinoma with anti-Hu paraneoplastic syndrome. N Eng J Med 2006;355:e4.
– reference: 9) Zaborowski MP, Michalak S. Cell-mediated immune responses in paraneoplastic neurological syndromes. Clin Dev Immunol 2013;630602:11.
– reference: 11) George J, Walter V, Peifer M, et al. Integrative genomic profiling of large-cell neuroendocrine carcinomas reveals distinct subtypes of high-grade neuroendocrine lung tumors. Nat Commun 2018;9:1048-1060.
– reference: 14) Derks JL, van Suylen RJ, Thunnissen E, et al. Chemotherapy for pulmonary large cell neuroendocrine carcinomas: does the regimen matter? Eur Respir J 2017;49:1601838.
– reference: 13) Ishikawa M, Kasamatsu T, Tsuda H, et al. Prognostic factors and optimal therapy for stage I-II neuroendocrine carcinomas of the uterine cervix: a multi-center retrospective study. Gynecol Oncol 2018;148:139-146.
– reference: 4) Ogura J, Adachi Y, Yasumoto K, et al. Large-cell neuro­endocrine carcinoma arising in the endometrium: a case report. Mol Clin Oncol 2018;8:575-578.
– reference: 6) West MS, Young BC. A case of confusion and bilateral temporal lesions in a young woman. Neurology 2010;75:67-68.
– reference: 12) Margolis B, Tergas AI, Chen L, et al. Natural history and outcome of neuroendocrine carcinoma of the cervix. Gynecol Oncol 2016;141:247-254.
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Snippet 症例は81歳女性．入院7か月前に子宮内膜原発大細胞神経内分泌癌（large cell neuroendocrine carcinoma; LCNEC）のため子宮全摘術および両側付属器切除術が施行された．入...
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SubjectTerms 傍腫瘍性神経症候群
子宮内膜原発大細胞神経内分泌癌
感覚性ニューロノパチー
抗Hu抗体
Title 子宮内膜原発大細胞神経内分泌癌に関連する抗Hu抗体陽性の傍腫瘍性sensory neuronopathyの1例
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Volume 60
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