Anti-pan-neurofascin IgG3 as a marker of fulminant autoimmune neuropathy

Neurol Neuroimmunol Neuroinflamm. 2019 Aug 16;6(5):e603. doi: 10.1212/NXI.0000000000000603. Print 2019 Sep.

Abstract

Objective: To identify and characterize patients with autoantibodies against different neurofascin (NF) isoforms.

Methods: Screening of a large cohort of patient sera for anti-NF autoantibodies by ELISA and further characterization by cell-based assays, epitope mapping, and complement binding assays.

Results: Two different clinical phenotypes became apparent in this study: The well-known clinical picture of subacute-onset severe sensorimotor neuropathy with tremor that is known to be associated with IgG4 autoantibodies against the paranodal isoform NF-155 was found in 2 patients. The second phenotype with a dramatic course of disease with tetraplegia and almost locked-in syndrome was associated with IgG3 autoantibodies against nodal and paranodal isoforms of NF in 3 patients. The epitope against which these autoantibodies were directed in this second phenotype was the common Ig domain found in all 3 NF isoforms. In contrast, anti-NF-155 IgG4 were directed against the NF-155-specific Fn3Fn4 domain. The description of a second phenotype of anti-NF-associated neuropathy is in line with some case reports of similar patients that were published in the last year.

Conclusions: Our results indicate that anti-pan-NF-associated neuropathy differs from anti-NF-155-associated neuropathy, and epitope and subclass play a major role in the pathogenesis and severity of anti-NF-associated neuropathy and should be determined to correctly classify patients, also in respect to possible differences in therapeutic response.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adolescent
  • Adult
  • Aged
  • Aged, 80 and over
  • Animals
  • Autoantibodies / blood*
  • Autoantibodies / immunology
  • Biomarkers / blood
  • Cell Adhesion Molecules / blood*
  • Cell Adhesion Molecules / immunology
  • Cell Line, Tumor
  • Child
  • Cohort Studies
  • Female
  • Guillain-Barre Syndrome / blood
  • Guillain-Barre Syndrome / diagnosis
  • Guillain-Barre Syndrome / immunology
  • HEK293 Cells
  • Humans
  • Immunoglobulin G / blood*
  • Immunoglobulin G / immunology
  • Male
  • Middle Aged
  • Nerve Growth Factors / blood*
  • Nerve Growth Factors / immunology
  • Polyradiculoneuropathy, Chronic Inflammatory Demyelinating / blood*
  • Polyradiculoneuropathy, Chronic Inflammatory Demyelinating / diagnosis*
  • Polyradiculoneuropathy, Chronic Inflammatory Demyelinating / immunology
  • Rats
  • Rats, Inbred Lew
  • Young Adult

Substances

  • Autoantibodies
  • Biomarkers
  • Cell Adhesion Molecules
  • Immunoglobulin G
  • NFASC protein, human
  • Nerve Growth Factors