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Clinical Autonomic Research
Published in: Clinical Autonomic Research 4/2009

01-08-2009 | Case Report

Autoimmune autonomic ganglionopathy: treatment by plasma exchanges and rituximab

Authors: Richard Imrich, Steven Vernino, Basil A. Eldadah, Courtney Holmes, David S. Goldstein

Published in: Clinical Autonomic Research | Issue 4/2009

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Abstract

This case report describes successful treatment of autoimmune autonomic ganglionopathy (AAG) in a 74-year-old woman by total plasma exchanges (PLEX) and rituximab. Two series of PLEX temporarily, but dramatically improved orthostatic intolerance and hypotension and baroreflex function, in a manner inversely related to ganglionic neuronal nicotinic receptor antibody titer. After rituximab treatment, the antibody titer was decreased modestly but persistently, and the patient had symptomatic and clinical laboratory evidence of continued benefit for at least 10 months, supporting the autoimmune pathogenesis of AAG.
Literature
1.
Goldstein DS, Holmes C, Dendi R, Li ST, Brentzel S, Vernino S (2002) Pandysautonomia associated with impaired ganglionic neurotransmission and circulating antibody to the neuronal nicotinic receptor. Clin Auton Res 12:281–285PubMedCrossRef
2.
Schroeder C, Vernino S, Birkenfeld AL, Tank J, Heusser K, Lipp A, Benter T, Lindschau C, Kettritz R, Luft FC, Jordan J (2005) Plasma exchange for primary autoimmune autonomic failure. N Engl J Med 353:1585–1590PubMedCrossRef
3.
Stubgen JP (2008) B cell-targeted therapy with rituximab and autoimmune neuromuscular disorders. J Neuroimmunol 204(1–2):1–12PubMedCrossRef
4.
Vernino S, Lindstrom J, Hopkins S, Wang Z, Low PA (2008) Characterization of ganglionic acetylcholine receptor autoantibodies. J Neuroimmunol 197:63–69PubMedCrossRef
5.
Vernino S, Low PA, Fealey RD, Stewart JD, Farrugia G, Lennon VA (2000) Autoantibodies to ganglionic acetylcholine receptors in autoimmune autonomic neuropathies. N Engl J Med 343:847–855PubMedCrossRef
Metadata
Title
Autoimmune autonomic ganglionopathy: treatment by plasma exchanges and rituximab
Authors
Richard Imrich
Steven Vernino
Basil A. Eldadah
Courtney Holmes
David S. Goldstein
Publication date
01-08-2009
Publisher
D. Steinkopff-Verlag
Published in
Clinical Autonomic Research / Issue 4/2009
Print ISSN: 0959-9851
Electronic ISSN: 1619-1560
DOI
https://doi.org/10.1007/s10286-009-0012-7

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