Characterization of IgG4 anti-neurofascin 155 antibody-positive polyneuropathy

Hidenori Ogata, Ryo Yamasaki, Hiwatashi Akio, Nobuyuki Oka, Nobutoshi Kawamura, Dai Matsuse, Motoi Kuwahara, Hidekazu Suzuki, Susumu Kusunoki, Yuichi Fujimoto, Koji Ikezoe, Hitaru Kishida, Fumiaki Tanaka, Takuya Matsushita, Hiroyuki Murai, Jun-Ichi Kira

研究成果: ジャーナルへの寄稿記事

50 引用 (Scopus)

抄録

Objective: To investigate anti-neurofascin 155 (NF155) antibody-positive chronic inflammatory demyelinating polyneuropathy (CIDP). Methods: Sera from 50 consecutive CIDP patients diagnosed in our clinic, 32 patients with multiple sclerosis, 40 patients with other neuropathies including 26 with Guillain–Barré syndrome (GBS)/Fisher syndrome, and 30 healthy controls were measured for anti-NF antibodies by flow cytometry using HEK293 cell lines stably expressing human NF155 or NF186. Four additional CIDP patients with anti-NF155 antibodies referred from other clinics were enrolled for clinical characterization. Results: The positivity rate for anti-NF155 antibodies in CIDP patients was 18% (9/50), who all showed a predominance of IgG4 subclass. No other subjects were positive, except one GBS patient harboring IgG1 anti-NF155 antibodies. No anti-NF155 antibody carriers had anti-NF186 antibodies. Anti-NF155 antibody-positive CIDP patients had a significantly younger onset age, higher frequency of drop foot, gait disturbance, tremor and distal acquired demyelinating symmetric phenotype, greater cervical root diameter on magnetic resonance imaging neurography, higher cerebrospinal fluid protein levels, and longer distal and F-wave latencies than anti-NF155 antibody-negative patients. Marked symmetric hypertrophy of cervical and lumbosacral roots/plexuses was present in all anti-NF155 antibody-positive CIDP patients examined by neurography. Biopsied sural nerves from two patients with anti-NF155 antibodies demonstrated subperineurial edema and occasional paranodal demyelination, but no vasculitis, inflammatory cell infiltrates, or onion bulbs. Among anti-NF155 antibody-positive patients, treatment responders more frequently had daily oral corticosteroids and/or immunosuppressants in addition to intravenous immunoglobulins than nonresponders did. Interpretation: Anti-NF155 antibodies occur in a subset of CIDP patients with distal-dominant involvement and symmetric nerve hypertrophy.

元の言語英語
ページ(範囲)960-971
ページ数12
ジャーナルAnnals of Clinical and Translational Neurology
2
発行部数10
DOI
出版物ステータス出版済み - 10 1 2015

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Polyneuropathies
Chronic Inflammatory Demyelinating Polyradiculoneuropathy
Immunoglobulin G
Antibodies
Hypertrophy
Anti-Idiotypic Antibodies
Miller Fisher Syndrome
Neurologic Gait Disorders
Lumbosacral Plexus
Cerebrospinal Fluid Proteins
Sural Nerve
Onions
HEK293 Cells
Intravenous Immunoglobulins
Demyelinating Diseases
Tremor
Immunosuppressive Agents
Vasculitis
Age of Onset
Multiple Sclerosis

All Science Journal Classification (ASJC) codes

  • Neuroscience(all)
  • Clinical Neurology

これを引用

Characterization of IgG4 anti-neurofascin 155 antibody-positive polyneuropathy. / Ogata, Hidenori; Yamasaki, Ryo; Akio, Hiwatashi; Oka, Nobuyuki; Kawamura, Nobutoshi; Matsuse, Dai; Kuwahara, Motoi; Suzuki, Hidekazu; Kusunoki, Susumu; Fujimoto, Yuichi; Ikezoe, Koji; Kishida, Hitaru; Tanaka, Fumiaki; Matsushita, Takuya; Murai, Hiroyuki; Kira, Jun-Ichi.

:: Annals of Clinical and Translational Neurology, 巻 2, 番号 10, 01.10.2015, p. 960-971.

研究成果: ジャーナルへの寄稿記事

Ogata, H, Yamasaki, R, Akio, H, Oka, N, Kawamura, N, Matsuse, D, Kuwahara, M, Suzuki, H, Kusunoki, S, Fujimoto, Y, Ikezoe, K, Kishida, H, Tanaka, F, Matsushita, T, Murai, H & Kira, J-I 2015, 'Characterization of IgG4 anti-neurofascin 155 antibody-positive polyneuropathy', Annals of Clinical and Translational Neurology, 巻. 2, 番号 10, pp. 960-971. https://doi.org/10.1002/acn3.248
Ogata, Hidenori ; Yamasaki, Ryo ; Akio, Hiwatashi ; Oka, Nobuyuki ; Kawamura, Nobutoshi ; Matsuse, Dai ; Kuwahara, Motoi ; Suzuki, Hidekazu ; Kusunoki, Susumu ; Fujimoto, Yuichi ; Ikezoe, Koji ; Kishida, Hitaru ; Tanaka, Fumiaki ; Matsushita, Takuya ; Murai, Hiroyuki ; Kira, Jun-Ichi. / Characterization of IgG4 anti-neurofascin 155 antibody-positive polyneuropathy. :: Annals of Clinical and Translational Neurology. 2015 ; 巻 2, 番号 10. pp. 960-971.
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abstract = "Objective: To investigate anti-neurofascin 155 (NF155) antibody-positive chronic inflammatory demyelinating polyneuropathy (CIDP). Methods: Sera from 50 consecutive CIDP patients diagnosed in our clinic, 32 patients with multiple sclerosis, 40 patients with other neuropathies including 26 with Guillain–Barr{\'e} syndrome (GBS)/Fisher syndrome, and 30 healthy controls were measured for anti-NF antibodies by flow cytometry using HEK293 cell lines stably expressing human NF155 or NF186. Four additional CIDP patients with anti-NF155 antibodies referred from other clinics were enrolled for clinical characterization. Results: The positivity rate for anti-NF155 antibodies in CIDP patients was 18{\%} (9/50), who all showed a predominance of IgG4 subclass. No other subjects were positive, except one GBS patient harboring IgG1 anti-NF155 antibodies. No anti-NF155 antibody carriers had anti-NF186 antibodies. Anti-NF155 antibody-positive CIDP patients had a significantly younger onset age, higher frequency of drop foot, gait disturbance, tremor and distal acquired demyelinating symmetric phenotype, greater cervical root diameter on magnetic resonance imaging neurography, higher cerebrospinal fluid protein levels, and longer distal and F-wave latencies than anti-NF155 antibody-negative patients. Marked symmetric hypertrophy of cervical and lumbosacral roots/plexuses was present in all anti-NF155 antibody-positive CIDP patients examined by neurography. Biopsied sural nerves from two patients with anti-NF155 antibodies demonstrated subperineurial edema and occasional paranodal demyelination, but no vasculitis, inflammatory cell infiltrates, or onion bulbs. Among anti-NF155 antibody-positive patients, treatment responders more frequently had daily oral corticosteroids and/or immunosuppressants in addition to intravenous immunoglobulins than nonresponders did. Interpretation: Anti-NF155 antibodies occur in a subset of CIDP patients with distal-dominant involvement and symmetric nerve hypertrophy.",
author = "Hidenori Ogata and Ryo Yamasaki and Hiwatashi Akio and Nobuyuki Oka and Nobutoshi Kawamura and Dai Matsuse and Motoi Kuwahara and Hidekazu Suzuki and Susumu Kusunoki and Yuichi Fujimoto and Koji Ikezoe and Hitaru Kishida and Fumiaki Tanaka and Takuya Matsushita and Hiroyuki Murai and Jun-Ichi Kira",
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T1 - Characterization of IgG4 anti-neurofascin 155 antibody-positive polyneuropathy

AU - Ogata, Hidenori

AU - Yamasaki, Ryo

AU - Akio, Hiwatashi

AU - Oka, Nobuyuki

AU - Kawamura, Nobutoshi

AU - Matsuse, Dai

AU - Kuwahara, Motoi

AU - Suzuki, Hidekazu

AU - Kusunoki, Susumu

AU - Fujimoto, Yuichi

AU - Ikezoe, Koji

AU - Kishida, Hitaru

AU - Tanaka, Fumiaki

AU - Matsushita, Takuya

AU - Murai, Hiroyuki

AU - Kira, Jun-Ichi

PY - 2015/10/1

Y1 - 2015/10/1

N2 - Objective: To investigate anti-neurofascin 155 (NF155) antibody-positive chronic inflammatory demyelinating polyneuropathy (CIDP). Methods: Sera from 50 consecutive CIDP patients diagnosed in our clinic, 32 patients with multiple sclerosis, 40 patients with other neuropathies including 26 with Guillain–Barré syndrome (GBS)/Fisher syndrome, and 30 healthy controls were measured for anti-NF antibodies by flow cytometry using HEK293 cell lines stably expressing human NF155 or NF186. Four additional CIDP patients with anti-NF155 antibodies referred from other clinics were enrolled for clinical characterization. Results: The positivity rate for anti-NF155 antibodies in CIDP patients was 18% (9/50), who all showed a predominance of IgG4 subclass. No other subjects were positive, except one GBS patient harboring IgG1 anti-NF155 antibodies. No anti-NF155 antibody carriers had anti-NF186 antibodies. Anti-NF155 antibody-positive CIDP patients had a significantly younger onset age, higher frequency of drop foot, gait disturbance, tremor and distal acquired demyelinating symmetric phenotype, greater cervical root diameter on magnetic resonance imaging neurography, higher cerebrospinal fluid protein levels, and longer distal and F-wave latencies than anti-NF155 antibody-negative patients. Marked symmetric hypertrophy of cervical and lumbosacral roots/plexuses was present in all anti-NF155 antibody-positive CIDP patients examined by neurography. Biopsied sural nerves from two patients with anti-NF155 antibodies demonstrated subperineurial edema and occasional paranodal demyelination, but no vasculitis, inflammatory cell infiltrates, or onion bulbs. Among anti-NF155 antibody-positive patients, treatment responders more frequently had daily oral corticosteroids and/or immunosuppressants in addition to intravenous immunoglobulins than nonresponders did. Interpretation: Anti-NF155 antibodies occur in a subset of CIDP patients with distal-dominant involvement and symmetric nerve hypertrophy.

AB - Objective: To investigate anti-neurofascin 155 (NF155) antibody-positive chronic inflammatory demyelinating polyneuropathy (CIDP). Methods: Sera from 50 consecutive CIDP patients diagnosed in our clinic, 32 patients with multiple sclerosis, 40 patients with other neuropathies including 26 with Guillain–Barré syndrome (GBS)/Fisher syndrome, and 30 healthy controls were measured for anti-NF antibodies by flow cytometry using HEK293 cell lines stably expressing human NF155 or NF186. Four additional CIDP patients with anti-NF155 antibodies referred from other clinics were enrolled for clinical characterization. Results: The positivity rate for anti-NF155 antibodies in CIDP patients was 18% (9/50), who all showed a predominance of IgG4 subclass. No other subjects were positive, except one GBS patient harboring IgG1 anti-NF155 antibodies. No anti-NF155 antibody carriers had anti-NF186 antibodies. Anti-NF155 antibody-positive CIDP patients had a significantly younger onset age, higher frequency of drop foot, gait disturbance, tremor and distal acquired demyelinating symmetric phenotype, greater cervical root diameter on magnetic resonance imaging neurography, higher cerebrospinal fluid protein levels, and longer distal and F-wave latencies than anti-NF155 antibody-negative patients. Marked symmetric hypertrophy of cervical and lumbosacral roots/plexuses was present in all anti-NF155 antibody-positive CIDP patients examined by neurography. Biopsied sural nerves from two patients with anti-NF155 antibodies demonstrated subperineurial edema and occasional paranodal demyelination, but no vasculitis, inflammatory cell infiltrates, or onion bulbs. Among anti-NF155 antibody-positive patients, treatment responders more frequently had daily oral corticosteroids and/or immunosuppressants in addition to intravenous immunoglobulins than nonresponders did. Interpretation: Anti-NF155 antibodies occur in a subset of CIDP patients with distal-dominant involvement and symmetric nerve hypertrophy.

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