AChR phosphorylation and indirect inhibition of AChR function in seronegative MG

Neurology. 2002 Dec 10;59(11):1682-8. doi: 10.1212/01.wnl.0000041625.41937.ff.

Abstract

Background: Approximately 10% to 20% of patients with autoimmune MG do not have antibodies to the acetylcholine receptor (AChR), so-called seronegative MG (SNMG). IgG antibodies from up to 70% of SNMG patients bind to the muscle-specific receptor tyrosine kinase, MuSK. The plasmas and non-IgG fractions from SNMG patients (and some with AChR antibodies) also contain a factor, perhaps an IgM antibody, that inhibits AChR function, but it is not clear how this factor acts and whether it is related to the MuSK IgG antibodies.

Methods: The authors studied 12 unselected SNMG plasmas and their non-IgG fractions; seven were positive for MuSK IgG antibodies. Ion flux assays, electrophysiology, phosphorylation, and kinase assays were used to look at mechanisms of action.

Results: Eight of the 12 plasmas and their non-IgG fractions inhibited AChR function, but the inhibitory activity was transient and did not correlate with the presence of MuSK IgG antibodies. Two of three plasmas added outside of a cell-attached patch pipette inhibited AChR function within the patch, and these two plasmas also increased AChR phosphorylation.

Conclusions: The authors propose that a plasma factor(s) in SNMG patients, distinct from MuSK IgG antibodies, binds to a muscle membrane receptor and activates a second messenger pathway leading to AChR phosphorylation and reduced AChR function. Identifying the target for this factor should lead to improved diagnosis of MG in MuSK antibody-negative patients and may provide new insights into the function of the neuromuscular junction and pathophysiological mechanisms in MG.

Publication types

  • Clinical Trial
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adenosine Triphosphate / metabolism
  • Adolescent
  • Adult
  • Aged
  • Autoantibodies / blood
  • Autoantibodies / immunology
  • Cell Line
  • Child
  • Child, Preschool
  • Cholinergic Antagonists / pharmacology*
  • Cyclic AMP-Dependent Protein Kinases / metabolism
  • Electrophysiology
  • Female
  • Humans
  • Immunoglobulin G / immunology
  • Immunoglobulin G / pharmacology
  • Infant
  • Male
  • Middle Aged
  • Myasthenia Gravis / blood*
  • Patch-Clamp Techniques
  • Phosphorylation
  • Protein Kinases / analysis
  • Receptor Protein-Tyrosine Kinases / blood*
  • Receptor Protein-Tyrosine Kinases / immunology
  • Receptors, Cholinergic / blood*
  • Receptors, Cholinergic / immunology
  • Receptors, Cholinergic / metabolism*
  • Sodium / metabolism

Substances

  • Autoantibodies
  • Cholinergic Antagonists
  • Immunoglobulin G
  • Receptors, Cholinergic
  • Adenosine Triphosphate
  • Sodium
  • Protein Kinases
  • MUSK protein, human
  • Receptor Protein-Tyrosine Kinases
  • Cyclic AMP-Dependent Protein Kinases