Evidence map›Paper›PMID 41052376›Full record

ArticleNeurology2025

Clinical, Electrophysiologic, and Pathologic Features of Anti-Contactin-Associated Protein 1 Autoimmune Nodopathy.

Takumi Tashiro, Hidenori Ogata, Yuki Fukami, Guzailiayi Maimaitijiang, Hitoshi Hayashida, Kazushi Deguchi, Yasuhiro Hamada, Yuko Ito, Masahiro Kanai, Atsuko Katsumoto and 30 more

Abstract read
In one paragraph

Article in Neurology, 2025. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 3 papers, 1 of them a synthesis that pooled it.

0numbers the graph read from it
0cells of the map it votes in
3citing papers in PubMed, 1 pooled it
–field-weighted citation impact
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

3 citing papers in PubMed, 1 synthesis or guideline pooled it.

  1. Pooled it
  2. Article
  3. Review
4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

40 authors.

Takumi TashiroDepartment of Neurology, Neurological Institute, Graduate School of Medical Sciences, Kyushu University, Fukuoka, Japan.
Hidenori OgataDepartment of Neurology, Neurological Institute, Graduate School of Medical Sciences, Kyushu University, Fukuoka, Japan.ORCID 0000-0001-8932-9731
Yuki FukamiDepartment of Neurology, Nagoya University Graduate School of Medicine, Japan.ORCID 0000-0003-1117-5925
Guzailiayi MaimaitijiangTranslational Neuroscience Research Center, Graduate School of Medicine, and School of Pharmacy at Fukuoka, International University of Health and Welfare, Okawa, Japan.
Hitoshi HayashidaDepartment of Neurology, Neurological Institute, Graduate School of Medical Sciences, Kyushu University, Fukuoka, Japan.
Kazushi DeguchiDepartment of Gastroenterology and Neurology, Graduate School of Medicine and Faculty of Medicine, Kagawa University, Japan.
Yasuhiro HamadaDepartment of Gastroenterology and Neurology, Graduate School of Medicine and Faculty of Medicine, Kagawa University, Japan.
Yuko ItoDepartment of Neurology, Saiseikai Matsuyama Hospital, Japan.
Masahiro KanaiDepartment of Neurology, Ichinomiya Nishi Hospital, Japan.
Atsuko KatsumotoDepartment of Neurology, National Center Hospital, National Center of Neurology and Psychiatry, Kodaira, Japan.
Ryota KishiDepartment of Neurology, Brain Research Institute, Niigata University, Japan.
Michiaki KogaSchool of Health Sciences, Yamaguchi University Graduate School of Medicine, Ube, Japan.ORCID 0000-0002-1356-955X
Takashi KurokawaDepartment of Neurology, Japan Community Healthcare Organization Tokyo Shinjuku Medical Center, Japan.
Akira MachidaDepartment of Neurology, Tsuchiura Kyodo General Hospital, Japan.ORCID 0000-0001-7457-8866
Masahito MiharaDepartment of Neurology, Kawasaki Medical School, Kurashiki, Japan.ORCID 0000-0001-5103-7784
Noriyuki MiyaueDepartment of Neurology, Saiseikai Matsuyama Hospital, Japan.
Yasuko MoriDepartment of Neurology, Gifu University Graduate School of Medicine, Japan.
Ichiro NabaDepartment of Neurology, Ikeda City Hospital, Japan.
Yoshiharu NakaeDepartment of Neurology, Saiseikai Yokohamashi Nanbu Hospital, Yokohama, Japan.
Akiko NakamuraDepartment of Neurology, Japan Community Healthcare Organization Tokyo Shinjuku Medical Center, Japan.
Hirotake NishimuraDepartment of Pathology, Kawasaki Medical School, Kurashiki, Japan.
Tomoko OkamotoDepartment of Neurology, National Center Hospital, National Center of Neurology and Psychiatry, Kodaira, Japan.ORCID 0000-0002-0983-3346
Etsuji SajiDepartment of Neurology, Brain Research Institute, Niigata University, Japan.
Ryota SatakeDepartment of Neurology, Kawasaki Medical School, Kurashiki, Japan.
Kenji SekiguchiDivision of Neurology, Kobe University Graduate School of Medicine, Japan.ORCID 0000-0003-4100-7316
Masahiro ShibuyaDepartment of Neurology, Saiseikai Yokohamashi Nanbu Hospital, Yokohama, Japan.
Manabu ShimodaDepartment of Neurology, Tottori Prefectural Central Hospital, Japan.
Takayoshi ShimohataDepartment of Neurology, Gifu University Graduate School of Medicine, Japan.ORCID 0000-0002-8788-9089
Yuki TakedaDivision of Neurology, Kobe University Graduate School of Medicine, Japan.
Kenta TanedaDepartment of Neurology, Tottori Prefectural Central Hospital, Japan.
Toshiyuki TezukaDepartment of Neurology, Niigata Prefectural Central Hospital, Joetsu, Japan.
Masaya TogoDivision of Neurology, Kobe University Graduate School of Medicine, Japan.
Takuya UeharaDepartment of Neurology, Ikeda City Hospital, Japan.
Keiji YamaguchiDepartment of Neurology, Ichinomiya Nishi Hospital, Japan.
Nobuaki YoshikuraDepartment of Neurology, Gifu University Graduate School of Medicine, Japan.
Ryo YamasakiDepartment of Neurology, Neurological Institute, Graduate School of Medical Sciences, Kyushu University, Fukuoka, Japan.ORCID 0000-0003-2071-7861
Jun-Ichi KiraTranslational Neuroscience Research Center, Graduate School of Medicine, and School of Pharmacy at Fukuoka, International University of Health and Welfare, Okawa, Japan.ORCID 0000-0001-5307-2671
Haruki KoikeDepartment of Neurology, Saga University, Japan; and.ORCID 0000-0002-4612-3183
Masahisa KatsunoDepartment of Neurology, Nagoya University Graduate School of Medicine, Japan.ORCID 0000-0001-9453-9311
Noriko IsobeDepartment of Neurology, Neurological Institute, Graduate School of Medical Sciences, Kyushu University, Fukuoka, Japan.ORCID 0000-0001-9525-4254

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

BACKGROUND AND

objectivesThe significance of anti-contactin-associated protein 1 (Caspr1) antibodies in autoimmune nodopathies (ANs) has not been fully established. The aim of this study was to elucidate the clinical profiles of Caspr1 AN.

methodsConsecutive serum samples were included from patients with chronic inflammatory demyelinating polyradiculoneuropathy (CIDP) nationwide who were referred to our laboratory for antibody testing with questionnaires as part of routine clinical practice and who fulfilled definite European Federation of Neurological Societies and Peripheral Nerve Society electrodiagnostic criteria. Anti-Caspr1 immunoglobulin (Ig) G was screened using in-house ELISA and confirmed using immunohistochemistry and Western blotting. Clinical data, electrophysiologic findings, and treatment responses were retrospectively collected. The pathologic features of sural nerves were also examined. Inflammatory Neuropathy Cause and Treatment (INCAT) disability scores, antibody titers, and serum neurofilament light chain (NfL) levels were analyzed at baseline and follow-up.

resultsOf 559 screened serum samples from patients with CIDP (median age at onset 54 years, 35% female), 19 cases with anti-Caspr1 IgG were identified; the main subclass was IgG4 (17 cases). Patients with IgG4 Caspr1 AN cases exhibited an older age at disease onset (median 67 years); male predominance (82%); high proportions of limb weakness (100%), gait disturbance (100%), tremor (65%), and sensory ataxia (82%); and very high CSF protein levels (249 mg/dL). Nerve conduction studies showed prolonged F-wave and distal motor latencies and reduced sensorimotor conduction velocities in all nerves tested. Light microscopy findings of sural nerve biopsy specimens from 4 patients with Caspr1 AN indicated a loss of myelinated fiber density and myelin ovoids without macrophage-mediated demyelination or onion bulbs. Electron microscopic evaluation demonstrated axo-glial detachment. Patients with IgG4 Caspr1 AN showed a poor response to IVIg (31%) and needed combined immunotherapies. Even after correcting for age, serum NfL levels were higher in patients with IgG4 Caspr1 AN than in healthy controls ( DISCUSSION: IgG4 Caspr1 AN presents with a similar clinical phenotype to other nodopathies (e.g., neurofascin 155 and contactin 1), but with an older age at onset. Changes in antibody titers may be a potential biomarker for monitoring disease activity.

Indexed as

Cell Adhesion Molecules, NeuronalPolyradiculoneuropathy, Chronic Inflammatory DemyelinatingSural NerveAgedAutoantibodiesElectrophysiologyFemaleHumansMaleMiddle AgedNerve Conduction StudiesNeurofilament ProteinsRetrospective StudiesAutoantibodiesCell Adhesion Molecules, NeuronalCNTNAP1 protein, humanneurofilament protein LNeurofilament Proteins

Identifiers

PMID41052376
PMCPMC12506924

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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.