抗Galactocerebroside抗体陽性の中枢末梢連合脱髄症にステロイドパルス療法が奏功した1例
59歳男性．複視・口渇で前医入院し糖尿病性ケトアシドーシスと診断．インスリン治療で血糖値改善後に四肢筋力低下・構音障害・嚥下障害・両上肢感覚障害が出現．C4～5，Th10髄内造影MRI病変，ミエリン塩基性蛋白高値，神経伝導速度で2領域以上の脱髄所見を認め，血清・髄液抗Neurofascin155抗体陰性ながら所見より中枢末梢連合脱髄症（Combined Central and Peripheral Demyelination; CCPD）と診断．ステロイドパルス療法後に脊髄病変は消失し退院．後に血清IgG抗Galactocerebroside抗体陽性を確認．CCPDは血漿交換やステロイド療法が...
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| Published in | 臨床神経学 Vol. 57; no. 12; pp. 747 - 752 |
|---|---|
| Main Authors | , , , , , , |
| Format | Journal Article |
| Language | Japanese |
| Published |
日本神経学会
2017
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| Subjects | |
| Online Access | Get full text |
| ISSN | 0009-918X 1882-0654 |
| DOI | 10.5692/clinicalneurol.cn-000977 |
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| Abstract | 59歳男性．複視・口渇で前医入院し糖尿病性ケトアシドーシスと診断．インスリン治療で血糖値改善後に四肢筋力低下・構音障害・嚥下障害・両上肢感覚障害が出現．C4～5，Th10髄内造影MRI病変，ミエリン塩基性蛋白高値，神経伝導速度で2領域以上の脱髄所見を認め，血清・髄液抗Neurofascin155抗体陰性ながら所見より中枢末梢連合脱髄症（Combined Central and Peripheral Demyelination; CCPD）と診断．ステロイドパルス療法後に脊髄病変は消失し退院．後に血清IgG抗Galactocerebroside抗体陽性を確認．CCPDは血漿交換やステロイド療法が有効だが，血清抗Galactocerebroside抗体陽性例の報告はなく症例の蓄積を要する． |
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| AbstractList | 59歳男性．複視・口渇で前医入院し糖尿病性ケトアシドーシスと診断．インスリン治療で血糖値改善後に四肢筋力低下・構音障害・嚥下障害・両上肢感覚障害が出現．C4～5，Th10髄内造影MRI病変，ミエリン塩基性蛋白高値，神経伝導速度で2領域以上の脱髄所見を認め，血清・髄液抗Neurofascin155抗体陰性ながら所見より中枢末梢連合脱髄症（Combined Central and Peripheral Demyelination; CCPD）と診断．ステロイドパルス療法後に脊髄病変は消失し退院．後に血清IgG抗Galactocerebroside抗体陽性を確認．CCPDは血漿交換やステロイド療法が有効だが，血清抗Galactocerebroside抗体陽性例の報告はなく症例の蓄積を要する． |
| Author | 鈴木, 祐 武藤, 多津郎 長谷川, 泰弘 島, さゆり 秋山, 久尚 星野, 俊 山田, 浩史 |
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| References | 1) Kamm C, Zettl UK. Autoimmune disorders affecting both the central and peripheral nervous system. Autoimmune Rev 2012;11:196-202. 15) Nishimura M, Saida T, Kuroki S, et al. Postinfectious encephalitis with anti-galactocerebroside antibody subsequent to Mycoplasma pneumoniae infection. J Neurol Sci 1996;140:91-95. 5) Butzkueven H, O’Brien TJ, Sedal L. Combined peripheral nerve and central nervous system demyelination in a patient with chronic inflammatory demyelinating polyneuropathy. J Clin Neurosci 1999;6:358-360. 12) Querol L, Nogales-Gadea G, Rojas-Garcia R, et al. Neurofascin IgG4 antibodies in CIDP associate with disabling tremor and poor response to IVIg. Neurology 2014;82:879-886. 10) Kriebel M, Wuchter J, Trinks S, et al. Neurofascin: a switch between neuronal plasticity and stability. Int J Biochem Cell Biol 2012;44:694-697. 11) Kawamura N, Yamasaki R, Yonekawa T, et al. Anti-neurofascin antibody in patients with combined central and peripheral demyelination. Neurology 2013;81:714-722. 16) 葛目大輔，佐島和晃，今野優子ら．抗ガラクトセレブロシド（Gal-Cer）IgG抗体陽性の急性散在性脳脊髄炎の1例．臨床神経 2015;55:550-554. 3) Rubin M, Karpati G, Carpenter S. Combined central and peripheral myelinopathy. Neurology 1987;37:1287-1290. 14) Saida K, Saida T, Brown MJ, et al. In vivo demyelination induced by intraneural injection of antigalactocerebroside serum. Am J Pathol 1979;95:99-116. 2) Zéphir H, Stojkovic T, Lacour A, et al. Relapsing demyelinating disease affecting both the central and peripheral nervous systems. J Neurol Neurosurg Psychiatry 2008;79:1032-1039. 13) Saida T, Saida K, Dorfman SH, et al. Experimental allergic neuritis induced by sensitization with galactocerebroside. Science 1979;204:1103-1106. 18) Katchanov J, Lünemann JD, Musuhr F, et al. Acute combined central and peripheral inflammatory demyelination. J Neurol Neurosurg Psychiatry 2004;75:1784-1786. 9) Ogata H, Matsuse D, Yamasaki R, et al. A nationwide survey of combined central and peripheral demyelination in Japan. J Neurol Neurosurg Psychiatry 2016;87:29-36. 7) Shima S, Kawamura N, Ishukawa T, et al. Anti-neutral glycolipid antibodies in encephalomyeloradiculoneuropathy. Neurology 2014;82:114-118. 8) Ueda A, Shima S, Miyashita T, et al. Anti-GM1 antibodies affect the integrity of lipid rafts. Mol Cell Neurosci 2010;45:355–362. 17) Samukawa M, Hirano M, Tsugawa J, et al. Refractory acute disseminated encephalomyelitis with anti-galactocerebroside antibody. Neurosci Res 2012;74:284-289. 4) Pareyson D, Ciano C, Flacchino F. et al. Combined central and peripheral acute demyelination. Ital J Neurol Sci 1993;14:83-86. 6) Kawamura N. Neurofascin: a novel target for combined central and peripheral demyelination. Clin Nuerol 2014;54:978-980. |
| References_xml | – reference: 7) Shima S, Kawamura N, Ishukawa T, et al. Anti-neutral glycolipid antibodies in encephalomyeloradiculoneuropathy. Neurology 2014;82:114-118. – reference: 16) 葛目大輔，佐島和晃，今野優子ら．抗ガラクトセレブロシド（Gal-Cer）IgG抗体陽性の急性散在性脳脊髄炎の1例．臨床神経 2015;55:550-554. – reference: 2) Zéphir H, Stojkovic T, Lacour A, et al. Relapsing demyelinating disease affecting both the central and peripheral nervous systems. J Neurol Neurosurg Psychiatry 2008;79:1032-1039. – reference: 17) Samukawa M, Hirano M, Tsugawa J, et al. Refractory acute disseminated encephalomyelitis with anti-galactocerebroside antibody. Neurosci Res 2012;74:284-289. – reference: 8) Ueda A, Shima S, Miyashita T, et al. Anti-GM1 antibodies affect the integrity of lipid rafts. Mol Cell Neurosci 2010;45:355–362. – reference: 11) Kawamura N, Yamasaki R, Yonekawa T, et al. Anti-neurofascin antibody in patients with combined central and peripheral demyelination. Neurology 2013;81:714-722. – reference: 15) Nishimura M, Saida T, Kuroki S, et al. Postinfectious encephalitis with anti-galactocerebroside antibody subsequent to Mycoplasma pneumoniae infection. J Neurol Sci 1996;140:91-95. – reference: 18) Katchanov J, Lünemann JD, Musuhr F, et al. Acute combined central and peripheral inflammatory demyelination. J Neurol Neurosurg Psychiatry 2004;75:1784-1786. – reference: 9) Ogata H, Matsuse D, Yamasaki R, et al. A nationwide survey of combined central and peripheral demyelination in Japan. J Neurol Neurosurg Psychiatry 2016;87:29-36. – reference: 12) Querol L, Nogales-Gadea G, Rojas-Garcia R, et al. Neurofascin IgG4 antibodies in CIDP associate with disabling tremor and poor response to IVIg. Neurology 2014;82:879-886. – reference: 14) Saida K, Saida T, Brown MJ, et al. In vivo demyelination induced by intraneural injection of antigalactocerebroside serum. Am J Pathol 1979;95:99-116. – reference: 1) Kamm C, Zettl UK. Autoimmune disorders affecting both the central and peripheral nervous system. Autoimmune Rev 2012;11:196-202. – reference: 3) Rubin M, Karpati G, Carpenter S. Combined central and peripheral myelinopathy. Neurology 1987;37:1287-1290. – reference: 4) Pareyson D, Ciano C, Flacchino F. et al. Combined central and peripheral acute demyelination. Ital J Neurol Sci 1993;14:83-86. – reference: 5) Butzkueven H, O’Brien TJ, Sedal L. Combined peripheral nerve and central nervous system demyelination in a patient with chronic inflammatory demyelinating polyneuropathy. J Clin Neurosci 1999;6:358-360. – reference: 10) Kriebel M, Wuchter J, Trinks S, et al. Neurofascin: a switch between neuronal plasticity and stability. Int J Biochem Cell Biol 2012;44:694-697. – reference: 13) Saida T, Saida K, Dorfman SH, et al. Experimental allergic neuritis induced by sensitization with galactocerebroside. Science 1979;204:1103-1106. – reference: 6) Kawamura N. Neurofascin: a novel target for combined central and peripheral demyelination. Clin Nuerol 2014;54:978-980. |
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| SubjectTerms | ステロイドパルス療法 中枢末梢連合脱髄症 抗Galactocerebroside抗体 抗Neurofascin抗体 |
| Title | 抗Galactocerebroside抗体陽性の中枢末梢連合脱髄症にステロイドパルス療法が奏功した1例 |
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