Anti-neurofascin antibody in patients with combined central and peripheral demyelination

Nobutoshi Kawamura, Ryo Yamasaki, Tomomi Yonekawa, Takuya Matsushita, Susumu Kusunoki, Shigemi Nagayama, Yasuo Fukuda, Hidenori Ogata, Dai Matsuse, Hiroyuki Murai, Jun-Ichi Kira

研究成果: ジャーナルへの寄稿記事

83 引用 (Scopus)

抄録

Objectives: We aimed to identify the target antigens for combined central and peripheral demyelination (CCPD). Methods: We screened target antigens by immunohistochemistry and immunoblotting using peripheral nerve tissues to identify target antigens recognized by serum antibodies from selected CCPD and chronic inflammatory demyelinating polyradiculoneuropathy (CIDP) cases. We then measured the level of antibody to the relevant antigen in 7 patients with CCPD, 16 patients with CIDP, 20 patients with multiple sclerosis, 20 patients with Guillain-Barré syndrome, 21 patients with other neuropathies, and 23 healthy controls (HC) by ELISA and cell-based assays using HEK293 cells. Results: At the initial screening, sera from 2 patients with CCPD showed cross-like binding to sciatic nerve sections at fixed intervals, with nearly perfect colocalization with neurofascin immunostaining at the node and paranode. ELISA with recombinant neurofascin revealed significantly higher mean optical density values in the CCPD group than in other disease groups and HC. Anti-neurofascin antibody positivity rates were 86% in patients with CCPD, 10% in patients with multiple sclerosis, 25% in patients with CIDP, 15% in patients with Guillain-Barré syndrome, and 0% in patients with other neuropathies and HC. The cell-based assay detected serum anti-neurofascin antibody in 5 of 7 patients with CCPD; all others were negative. CSF samples examined from 2 patients with CCPD were both positive. In anti-neurofascin antibody- positive CCPD patients, including those with a limited response to corticosteroids, IV immunoglobulin or plasma exchange alleviated the symptoms. Conclusion: Anti-neurofascin antibody is frequently present in patients with CCPD. Recognition of this antibody may be important, because patients with CCPD who are antibody positive respond well to IV immunoglobulin or plasma exchange.

元の言語英語
ページ(範囲)714-722
ページ数9
ジャーナルNeurology
81
発行部数8
DOI
出版物ステータス出版済み - 8 20 2013

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Demyelinating Diseases
Anti-Idiotypic Antibodies
Chronic Inflammatory Demyelinating Polyradiculoneuropathy
Antigens
Plasma Exchange
Antibodies
Intravenous Immunoglobulins
Multiple Sclerosis
Serum
Enzyme-Linked Immunosorbent Assay
Nerve Tissue
HEK293 Cells
Sciatic Nerve
Peripheral Nerves
Immunoblotting

All Science Journal Classification (ASJC) codes

  • Clinical Neurology

これを引用

Anti-neurofascin antibody in patients with combined central and peripheral demyelination. / Kawamura, Nobutoshi; Yamasaki, Ryo; Yonekawa, Tomomi; Matsushita, Takuya; Kusunoki, Susumu; Nagayama, Shigemi; Fukuda, Yasuo; Ogata, Hidenori; Matsuse, Dai; Murai, Hiroyuki; Kira, Jun-Ichi.

:: Neurology, 巻 81, 番号 8, 20.08.2013, p. 714-722.

研究成果: ジャーナルへの寄稿記事

Kawamura, Nobutoshi ; Yamasaki, Ryo ; Yonekawa, Tomomi ; Matsushita, Takuya ; Kusunoki, Susumu ; Nagayama, Shigemi ; Fukuda, Yasuo ; Ogata, Hidenori ; Matsuse, Dai ; Murai, Hiroyuki ; Kira, Jun-Ichi. / Anti-neurofascin antibody in patients with combined central and peripheral demyelination. :: Neurology. 2013 ; 巻 81, 番号 8. pp. 714-722.
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title = "Anti-neurofascin antibody in patients with combined central and peripheral demyelination",
abstract = "Objectives: We aimed to identify the target antigens for combined central and peripheral demyelination (CCPD). Methods: We screened target antigens by immunohistochemistry and immunoblotting using peripheral nerve tissues to identify target antigens recognized by serum antibodies from selected CCPD and chronic inflammatory demyelinating polyradiculoneuropathy (CIDP) cases. We then measured the level of antibody to the relevant antigen in 7 patients with CCPD, 16 patients with CIDP, 20 patients with multiple sclerosis, 20 patients with Guillain-Barr{\'e} syndrome, 21 patients with other neuropathies, and 23 healthy controls (HC) by ELISA and cell-based assays using HEK293 cells. Results: At the initial screening, sera from 2 patients with CCPD showed cross-like binding to sciatic nerve sections at fixed intervals, with nearly perfect colocalization with neurofascin immunostaining at the node and paranode. ELISA with recombinant neurofascin revealed significantly higher mean optical density values in the CCPD group than in other disease groups and HC. Anti-neurofascin antibody positivity rates were 86{\%} in patients with CCPD, 10{\%} in patients with multiple sclerosis, 25{\%} in patients with CIDP, 15{\%} in patients with Guillain-Barr{\'e} syndrome, and 0{\%} in patients with other neuropathies and HC. The cell-based assay detected serum anti-neurofascin antibody in 5 of 7 patients with CCPD; all others were negative. CSF samples examined from 2 patients with CCPD were both positive. In anti-neurofascin antibody- positive CCPD patients, including those with a limited response to corticosteroids, IV immunoglobulin or plasma exchange alleviated the symptoms. Conclusion: Anti-neurofascin antibody is frequently present in patients with CCPD. Recognition of this antibody may be important, because patients with CCPD who are antibody positive respond well to IV immunoglobulin or plasma exchange.",
author = "Nobutoshi Kawamura and Ryo Yamasaki and Tomomi Yonekawa and Takuya Matsushita and Susumu Kusunoki and Shigemi Nagayama and Yasuo Fukuda and Hidenori Ogata and Dai Matsuse and Hiroyuki Murai and Jun-Ichi Kira",
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T1 - Anti-neurofascin antibody in patients with combined central and peripheral demyelination

AU - Kawamura, Nobutoshi

AU - Yamasaki, Ryo

AU - Yonekawa, Tomomi

AU - Matsushita, Takuya

AU - Kusunoki, Susumu

AU - Nagayama, Shigemi

AU - Fukuda, Yasuo

AU - Ogata, Hidenori

AU - Matsuse, Dai

AU - Murai, Hiroyuki

AU - Kira, Jun-Ichi

PY - 2013/8/20

Y1 - 2013/8/20

N2 - Objectives: We aimed to identify the target antigens for combined central and peripheral demyelination (CCPD). Methods: We screened target antigens by immunohistochemistry and immunoblotting using peripheral nerve tissues to identify target antigens recognized by serum antibodies from selected CCPD and chronic inflammatory demyelinating polyradiculoneuropathy (CIDP) cases. We then measured the level of antibody to the relevant antigen in 7 patients with CCPD, 16 patients with CIDP, 20 patients with multiple sclerosis, 20 patients with Guillain-Barré syndrome, 21 patients with other neuropathies, and 23 healthy controls (HC) by ELISA and cell-based assays using HEK293 cells. Results: At the initial screening, sera from 2 patients with CCPD showed cross-like binding to sciatic nerve sections at fixed intervals, with nearly perfect colocalization with neurofascin immunostaining at the node and paranode. ELISA with recombinant neurofascin revealed significantly higher mean optical density values in the CCPD group than in other disease groups and HC. Anti-neurofascin antibody positivity rates were 86% in patients with CCPD, 10% in patients with multiple sclerosis, 25% in patients with CIDP, 15% in patients with Guillain-Barré syndrome, and 0% in patients with other neuropathies and HC. The cell-based assay detected serum anti-neurofascin antibody in 5 of 7 patients with CCPD; all others were negative. CSF samples examined from 2 patients with CCPD were both positive. In anti-neurofascin antibody- positive CCPD patients, including those with a limited response to corticosteroids, IV immunoglobulin or plasma exchange alleviated the symptoms. Conclusion: Anti-neurofascin antibody is frequently present in patients with CCPD. Recognition of this antibody may be important, because patients with CCPD who are antibody positive respond well to IV immunoglobulin or plasma exchange.

AB - Objectives: We aimed to identify the target antigens for combined central and peripheral demyelination (CCPD). Methods: We screened target antigens by immunohistochemistry and immunoblotting using peripheral nerve tissues to identify target antigens recognized by serum antibodies from selected CCPD and chronic inflammatory demyelinating polyradiculoneuropathy (CIDP) cases. We then measured the level of antibody to the relevant antigen in 7 patients with CCPD, 16 patients with CIDP, 20 patients with multiple sclerosis, 20 patients with Guillain-Barré syndrome, 21 patients with other neuropathies, and 23 healthy controls (HC) by ELISA and cell-based assays using HEK293 cells. Results: At the initial screening, sera from 2 patients with CCPD showed cross-like binding to sciatic nerve sections at fixed intervals, with nearly perfect colocalization with neurofascin immunostaining at the node and paranode. ELISA with recombinant neurofascin revealed significantly higher mean optical density values in the CCPD group than in other disease groups and HC. Anti-neurofascin antibody positivity rates were 86% in patients with CCPD, 10% in patients with multiple sclerosis, 25% in patients with CIDP, 15% in patients with Guillain-Barré syndrome, and 0% in patients with other neuropathies and HC. The cell-based assay detected serum anti-neurofascin antibody in 5 of 7 patients with CCPD; all others were negative. CSF samples examined from 2 patients with CCPD were both positive. In anti-neurofascin antibody- positive CCPD patients, including those with a limited response to corticosteroids, IV immunoglobulin or plasma exchange alleviated the symptoms. Conclusion: Anti-neurofascin antibody is frequently present in patients with CCPD. Recognition of this antibody may be important, because patients with CCPD who are antibody positive respond well to IV immunoglobulin or plasma exchange.

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