Literature DB >> 31924415

A comprehensive analysis of the clinical characteristics and laboratory features in 179 patients with autoimmune autonomic ganglionopathy.

Shunya Nakane1, Akihiro Mukaino2, Osamu Higuchi3, Maeda Yasuhiro4, Koutaro Takamatsu5, Makoto Yamakawa6, Mari Watari7, Nozomu Tawara8, Kei-Ichi Nakahara9, Atsushi Kawakami10, Hidenori Matsuo11, Yukio Ando12.   

Abstract

The clinical importance of autoantibodies against the ganglionic acetylcholine receptor (gAChR) remains to be fully elucidated. We aimed to identify the clinical characteristics of autoimmune autonomic ganglionopathy (AAG) in patients with gAChR autoantibodies. For this cohort investigation, serum samples were obtained from patients with AAG between 2012 and 2018 in Japan. We measured the levels of autoantibodies against gAChRα3 and gAChRβ4 and evaluated clinical features, as well as assessing the laboratory investigation results among the included patients. A total of 179 patients tested positive for antibodies, including 116 gAChRα3-positive, 13 gAChRβ4-positive, and 50 double antibody-positive patients. Seropositive AAG patients exhibited widespread autonomic dysfunction. Extra-autonomic manifestations including sensory disturbance, central nervous system involvement, endocrine disorders, autoimmune diseases, and tumours were present in 118 patients (83%). We observed significant differences in the frequencies of several autonomic and extra-autonomic symptoms among the three groups. Our 123I-metaiodobenzylguanidine myocardial scintigraphy analysis of the entire cohort revealed that the heart-to-mediastinum ratio had decreased by 80%. The present study is the first to demonstrate that patients with AAG who are seropositive for anti-gAChRβ4 autoantibodies exhibit unique autonomic and extra-autonomic signs. Decreased cardiac uptake occurred in most cases, indicating that 123I- metaiodobenzylguanidine myocardial scintigraphy may be useful for monitoring AAG. Therefore, our findings indicate that gAChRα3 and gAChRβ4 autoantibodies cause functional changes in postganglionic fibres in the autonomic nervous system and extra-autonomic manifestations in seropositive patients with AAG.
Copyright © 2020 Elsevier Ltd. All rights reserved.

Entities:  

Keywords:  Anti-ganglionic acetylcholine receptor (gAChR) antibodies; Autoantibody: (123)I- metaiodobenzylguanidine (MIBG) myocardial scintigraphy; Autoimmune autonomic ganglionopathy (AAG); Autonomic dysfunction

Mesh:

Substances:

Year:  2020        PMID: 31924415     DOI: 10.1016/j.jaut.2020.102403

Source DB:  PubMed          Journal:  J Autoimmun        ISSN: 0896-8411            Impact factor:   7.094


  5 in total

1.  gAChR antibodies in children and adolescents with acquired autoimmune dysautonomia in Japan.

Authors:  Makoto Yamakawa; Mari Watari; Ken-Ichi Torii; Ichiro Kuki; Masashi Miharu; Momoko Kawazu; Akihiro Mukaino; Osamu Higuchi; Yasuhiro Maeda; Tokunori Ikeda; Koutaro Takamatsu; Nozomu Tawara; Keiichi Nakahara; Hidenori Matsuo; Mitsuharu Ueda; Takao Takahashi; Shunya Nakane
Journal:  Ann Clin Transl Neurol       Date:  2021-02-23       Impact factor: 4.511

2.  Autoimmune Autonomic Ganglionopathy Presenting as Constipation.

Authors:  Abdelwahab Ahmed; Shannon Lohman; Suraj Suresh; Abdullah Naji; Sarah Russell; Eva Alsheik; Keith Mullins
Journal:  Cureus       Date:  2022-02-10

3.  Effectiveness of treatment for 31 patients with seropositive autoimmune autonomic ganglionopathy in Japan.

Authors:  Toshiyuki Hayashi; Shunya Nakane; Akihiro Mukaino; Osamu Higuchi; Makoto Yamakawa; Hidenori Matsuo; Kazumi Kimura
Journal:  Ther Adv Neurol Disord       Date:  2022-08-03       Impact factor: 6.430

4.  Progressive Encephalomyelitis with Rigidity and Myoclonus (PERM)-like Symptoms Associated with Anti-ganglionic Acetylcholine Receptor Antibodies.

Authors:  Yuki Kitazaki; Masamichi Ikawa; Toru Kishitani; Tomoko Kamisawa; Shunya Nakane; Yasunari Nakamoto; Tadanori Hamano
Journal:  Intern Med       Date:  2021-02-15       Impact factor: 1.271

5.  Association between neurosarcoidosis with autonomic dysfunction and anti-ganglionic acetylcholine receptor antibodies.

Authors:  Makoto Oishi; Akihiro Mukaino; Misako Kunii; Asami Saito; Yukimasa Arita; Haruki Koike; Osamu Higuchi; Yasuhiro Maeda; Norio Abiru; Naohiro Yamaguchi; Hiroaki Kawano; Eiko Tsuiki; Tomonori Tanaka; Hidenori Matsuo; Masahisa Katsuno; Fumiaki Tanaka; Akira Tsujino; Shunya Nakane
Journal:  J Neurol       Date:  2021-04-21       Impact factor: 4.849

  5 in total

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