ELectron microscopic abnormality and therapeutic efficacy in chronic inflammatory demyelinating polyneuropathy with anti-neurofascin155 immunoglobulin G4 antibody

Motoi Kuwahara; Hidekazu Suzuki; Nobuyuki Oka; Hidenori Ogata; Satoshi Yanagimoto; Shuji Sadakane; Yuta Fukumoto; Masaki Yamana; Yoshiko Yuhara; Keisuke Yoshikawa; Miyuki Morikawa; Shigeru Kawai; Masahiro Okazaki; Toru Tsujimoto; Jun Ichi Kira; Susumu Kusunoki

doi:10.1002/mus.25757

ELectron microscopic abnormality and therapeutic efficacy in chronic inflammatory demyelinating polyneuropathy with anti-neurofascin155 immunoglobulin G4 antibody

Motoi Kuwahara, Hidekazu Suzuki, Nobuyuki Oka, Hidenori Ogata, Satoshi Yanagimoto, Shuji Sadakane, Yuta Fukumoto, Masaki Yamana, Yoshiko Yuhara, Keisuke Yoshikawa, Miyuki Morikawa, Shigeru Kawai, Masahiro Okazaki, Toru Tsujimoto, Jun Ichi Kira, Susumu Kusunoki

Department of Neurogy

Research output: Contribution to journal › Article › peer-review

13 Citations (Scopus)

Abstract

Introduction: Neurofascin155 (NF155) is a target antigen for autoantibodies in a subset of chronic inflammatory demyelinating polyneuropathy (CIDP). Methods: We report the cases of 4 patients with anti-NF155 immunoglobulin G4 (IgG4) antibody-positive CIDP who underwent sural nerve biopsies. Results: All patients were relatively young at onset. Three patients experienced tremors, and 2 patients had severe ataxia. Although the response to intravenous immunoglobulin was poor in all patients, plasma exchange and corticosteroids were at least partially effective. Immunoadsorption plasmapheresis was performed in 1 patient but was ineffective. Electron microscopic examination of sural nerve biopsies revealed loss of paranodal transverse bands in all patients. Discussion: Anti-NF155 IgG4 antibody-positive CIDP shows distinctive clinicopathological features, indicating that the IgG4 antibody is directly associated with the pathogenic mechanisms of anti-NF155 IgG4 antibody-positive CIDP. Muscle Nerve 57: 498–502, 2018.

Original language	English
Pages (from-to)	498-502
Number of pages	5
Journal	Muscle and Nerve
Volume	57
Issue number	3
DOIs	https://doi.org/10.1002/mus.25757
Publication status	Published - Mar 2018

All Science Journal Classification (ASJC) codes

Physiology
Clinical Neurology
Cellular and Molecular Neuroscience
Physiology (medical)

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Kuwahara, M., Suzuki, H., Oka, N., Ogata, H., Yanagimoto, S., Sadakane, S., Fukumoto, Y., Yamana, M., Yuhara, Y., Yoshikawa, K., Morikawa, M., Kawai, S., Okazaki, M., Tsujimoto, T., Kira, J. I., & Kusunoki, S. (2018). ELectron microscopic abnormality and therapeutic efficacy in chronic inflammatory demyelinating polyneuropathy with anti-neurofascin155 immunoglobulin G4 antibody. Muscle and Nerve, 57(3), 498-502. https://doi.org/10.1002/mus.25757

ELectron microscopic abnormality and therapeutic efficacy in chronic inflammatory demyelinating polyneuropathy with anti-neurofascin155 immunoglobulin G4 antibody. / Kuwahara, Motoi; Suzuki, Hidekazu; Oka, Nobuyuki; Ogata, Hidenori; Yanagimoto, Satoshi; Sadakane, Shuji; Fukumoto, Yuta; Yamana, Masaki; Yuhara, Yoshiko; Yoshikawa, Keisuke; Morikawa, Miyuki; Kawai, Shigeru; Okazaki, Masahiro; Tsujimoto, Toru; Kira, Jun Ichi; Kusunoki, Susumu.

In: Muscle and Nerve, Vol. 57, No. 3, 03.2018, p. 498-502.

Research output: Contribution to journal › Article › peer-review

Kuwahara, M, Suzuki, H, Oka, N, Ogata, H, Yanagimoto, S, Sadakane, S, Fukumoto, Y, Yamana, M, Yuhara, Y, Yoshikawa, K, Morikawa, M, Kawai, S, Okazaki, M, Tsujimoto, T, Kira, JI & Kusunoki, S 2018, 'ELectron microscopic abnormality and therapeutic efficacy in chronic inflammatory demyelinating polyneuropathy with anti-neurofascin155 immunoglobulin G4 antibody', Muscle and Nerve, vol. 57, no. 3, pp. 498-502. https://doi.org/10.1002/mus.25757

Kuwahara, Motoi ; Suzuki, Hidekazu ; Oka, Nobuyuki ; Ogata, Hidenori ; Yanagimoto, Satoshi ; Sadakane, Shuji ; Fukumoto, Yuta ; Yamana, Masaki ; Yuhara, Yoshiko ; Yoshikawa, Keisuke ; Morikawa, Miyuki ; Kawai, Shigeru ; Okazaki, Masahiro ; Tsujimoto, Toru ; Kira, Jun Ichi ; Kusunoki, Susumu. / ELectron microscopic abnormality and therapeutic efficacy in chronic inflammatory demyelinating polyneuropathy with anti-neurofascin155 immunoglobulin G4 antibody. In: Muscle and Nerve. 2018 ; Vol. 57, No. 3. pp. 498-502.

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abstract = "Introduction: Neurofascin155 (NF155) is a target antigen for autoantibodies in a subset of chronic inflammatory demyelinating polyneuropathy (CIDP). Methods: We report the cases of 4 patients with anti-NF155 immunoglobulin G4 (IgG4) antibody-positive CIDP who underwent sural nerve biopsies. Results: All patients were relatively young at onset. Three patients experienced tremors, and 2 patients had severe ataxia. Although the response to intravenous immunoglobulin was poor in all patients, plasma exchange and corticosteroids were at least partially effective. Immunoadsorption plasmapheresis was performed in 1 patient but was ineffective. Electron microscopic examination of sural nerve biopsies revealed loss of paranodal transverse bands in all patients. Discussion: Anti-NF155 IgG4 antibody-positive CIDP shows distinctive clinicopathological features, indicating that the IgG4 antibody is directly associated with the pathogenic mechanisms of anti-NF155 IgG4 antibody-positive CIDP. Muscle Nerve 57: 498–502, 2018.",

author = "Motoi Kuwahara and Hidekazu Suzuki and Nobuyuki Oka and Hidenori Ogata and Satoshi Yanagimoto and Shuji Sadakane and Yuta Fukumoto and Masaki Yamana and Yoshiko Yuhara and Keisuke Yoshikawa and Miyuki Morikawa and Shigeru Kawai and Masahiro Okazaki and Toru Tsujimoto and Kira, {Jun Ichi} and Susumu Kusunoki",

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AU - Sadakane, Shuji

AU - Fukumoto, Yuta

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AU - Morikawa, Miyuki

AU - Kawai, Shigeru

AU - Okazaki, Masahiro

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