Case report: Recurring potassium channel complex autoimmunity-related neuropathic pain

Front Immunol. 2024 Oct 25:15:1390171. doi: 10.3389/fimmu.2024.1390171. eCollection 2024.

Abstract

Voltage-gated potassium channel (VGKC) complex autoimmunity associated with nerve hyperexcitability is an uncommon clinical spectrum. It is mostly characterized by limbic encephalitis, continuous neuromyotonia, and dysautonomia. Pain, however, has rarely been reported as the first symptom. The present study describes a case of persistent neuropathic pain as the only symptom associated with a positive serum contactin-associated protein-like 2 (CASPR2) auto-antibody in a 41-year-old female patient. Her pain was completely relieved with steroids and intravenous immunoglobulin (IVIG) therapy. Nevertheless, the pain recurred 1 year later, consistent with an immunofluorescence titer of the CASPR2 antibody. Our case shows that neuropathic pain may occur as the first and only manifestation of a VGKC complex autoimmunity disorder. VGKC antibody titers might be an indication of pain severity. Steroids coupled with IVIG are effective, but relapse may still occur.

Keywords: antibody titer; contactin-associated protein-like 2; neuropathic pain; recurrence; voltage-gated potassium channel.

Publication types

  • Case Reports

MeSH terms

  • Adult
  • Autoantibodies* / blood
  • Autoantibodies* / immunology
  • Autoimmunity*
  • Female
  • Humans
  • Immunoglobulins, Intravenous / therapeutic use
  • Membrane Proteins / immunology
  • Nerve Tissue Proteins* / immunology
  • Neuralgia* / etiology
  • Neuralgia* / immunology
  • Potassium Channels, Voltage-Gated* / immunology
  • Recurrence

Substances

  • Potassium Channels, Voltage-Gated
  • Autoantibodies
  • Nerve Tissue Proteins
  • CNTNAP2 protein, human
  • Membrane Proteins
  • Immunoglobulins, Intravenous

Grants and funding

The author(s) declare that financial support was received for the research, authorship, and/or publication of this article. This study was supported in part by Jiangsu Province Hospital (the First Affiliated Hospital with Nanjing Medical University) Clinical Capacity Enhancement Project in 2020 (grant number JSPH-MC-2020-9) and National Natural Science Foundation of China (grant number 82371283).