Literature DB >> 20837310

Isolated sympathetic failure with autoimmune autonomic ganglionopathy.

Philip R Fischer1, Paola Sandroni, Sean J Pittock, Co-burn J Porter, Lenora M Lehwald, Satish R Raj.   

Abstract

A 16-year-old boy had a gradual onset of post-exercise myalgia with progressive fatigue and dizziness. He had bradycardia (37 beats/minute) with low supine and normal standing norepinephrine levels (56 and 311 pg/mL, respectively). He had absent sympathetically mediated vasoconstrictor responses during Valsalva maneuver testing. Circulating ganglionic acetylcholine receptor antibodies were identified. Response was gradual to treatment with intravenous immunoglobulin combined with aggressive symptomatic interventions (permanent pacemaker implantation and treatment with pyridostigmine, midodrine, and modafinil). After the intravenous immunoglobulin treatment, his autoantibody levels decreased and the autonomic abnormalities resolved. After a reconditioning exercise program and eventually undetectable antibody titers, he achieved complete recovery. The patient continued to do well after his pacemaker was removed and his medications were discontinued. Thus, severe isolated sympathetic nervous system failure can occur in adolescents with autoimmune autonomic ganglionopathy, and multifaceted treatment can be effective.
Copyright © 2010 Elsevier Inc. All rights reserved.

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Year:  2010        PMID: 20837310     DOI: 10.1016/j.pediatrneurol.2010.05.003

Source DB:  PubMed          Journal:  Pediatr Neurol        ISSN: 0887-8994            Impact factor:   3.372


  4 in total

1.  The spectrum of clinicopathological features in pure autonomic neuropathy.

Authors:  Haruki Koike; Rina Hashimoto; Minoru Tomita; Yuichi Kawagashira; Masahiro Iijima; Shigeru Koyano; Takayuki Momoo; Hiroyuki Yuasa; Shigehisa Mitake; Mana Higashihara; Kenichi Kaida; Daisuke Yamamoto; Shin Hisahara; Shun Shimohama; Yoshiharu Nakae; Ken Johkura; Steven Vernino; Gen Sobue
Journal:  J Neurol       Date:  2012-02-24       Impact factor: 4.849

2.  Ganglionic Antibody Level as a Predictor of Severity of Autonomic Failure.

Authors:  Jeremy K Cutsforth-Gregory; Andrew McKeon; Elizabeth A Coon; David M Sletten; Mariana Suarez; Paola Sandroni; Wolfgang Singer; Eduardo E Benarroch; Robert D Fealey; Phillip A Low
Journal:  Mayo Clin Proc       Date:  2018-08-28       Impact factor: 7.616

3.  Cognitive impairments associated with CFS and POTS.

Authors:  Lindzi Shanks; Leonard A Jason; Meredyth Evans; Abigail Brown
Journal:  Front Physiol       Date:  2013-05-16       Impact factor: 4.566

4.  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

  4 in total

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