Literature DB >> 28214088

A case of seropositive autoimmune autonomic ganglionopathy with diffuse esophageal spasm.

Nobutoshi Morimoto1, Sakuma Takahashi2, Tomoki Inaba2, Motonori Takamiya3, Yasuhiko Kageyama3, Mizuki Morimoto3, Yoshiaki Takahashi4, Hirotake Nishimura5, Shunya Nakane6, Koji Abe7.   

Abstract

Autoimmune autonomic ganglionopathy (AAG) is an immune-mediated disorder that leads to various autonomic failures associated with anti-ganglionic acetylcholine receptor antibodies (anti-gAChR-Abs). Diffuse esophageal spasm (DES) is an uncommon esophageal motility disorder. We herein report the case of a 68-year-old woman with DES as a partial symptom of AAG. She presented with chronic esophageal transit failure, constipation, and numbness of the hands and feet, Adie's pupil, thermal hypoalgesia, and decreased deep tendon reflexes. Right sural nerve biopsy showed significantly decreased numbers of small myelinated fibers. Barium swallowing X-ray showed repetitive simultaneous contractions indicating DES in the esophagus. Gastrointestinal endoscopy and CT image showed a dilated esophageal lumen and liquid effusion. Simultaneously, serum anti-gAChR-α3-Ab indicating AAG was detected. After pulse intravenous methylprednisolone (IVMP) and intravenous immunoglobulin therapy (IVIg), the bolus progression and liquid effusion improved, suggesting that DES is an important gastrointestinal symptom of AAG.
Copyright © 2017 Elsevier Ltd. All rights reserved.

Entities:  

Keywords:  Anti-ganglionic acetylcholine receptor α3 antibody; Autoimmune autonomic ganglionopathy (AAG); Diffuse esophageal spasm (DES); Intravenous immunoglobulin therapy (IVIg)

Mesh:

Substances:

Year:  2017        PMID: 28214088     DOI: 10.1016/j.jocn.2017.01.027

Source DB:  PubMed          Journal:  J Clin Neurosci        ISSN: 0967-5868            Impact factor:   1.961


  4 in total

1.  Anti-ganglionic AChR antibodies in Japanese patients with motility disorders.

Authors:  Akihiro Mukaino; Hitomi Minami; Hajime Isomoto; Hitomi Hamamoto; Eikichi Ihara; Yasuhiro Maeda; Osamu Higuchi; Tohru Okanishi; Yohei Kokudo; Kazushi Deguchi; Fumisato Sasaki; Toshihito Ueki; Ken-Ya Murata; Takeshi Yoshida; Mistuyo Kinjo; Yoshihiro Ogawa; Akio Ido; Hidenori Matsuo; Kazuhiko Nakao; Shunya Nakane
Journal:  J Gastroenterol       Date:  2018-05-15       Impact factor: 7.527

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

Review 3.  Adie's Pupil: A Diagnostic Challenge for the Physician.

Authors:  Sui-Yi Xu; Mao-Mei Song; Ling Li; Chang-Xin Li
Journal:  Med Sci Monit       Date:  2022-03-08

4.  Detecting gastrointestinal manifestations in patients with systemic sclerosis using anti-gAChR antibodies.

Authors:  Shunya Nakane; Masataka Umeda; Shin-Ya Kawashiri; Akihiro Mukaino; Kunihiro Ichinose; Osamu Higuchi; Yasuhiro Maeda; Hideki Nakamura; Hidenori Matsuo; Atsushi Kawakami
Journal:  Arthritis Res Ther       Date:  2020-02-21       Impact factor: 5.156

  4 in total

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