An Atypical Phenotype of Chronic Inflammatory Demyelinating Polyradiculoneuropathy Associated with Ocular Palsy, IgM-anti Ganglioside Antibody, and Fever-induced Recurrence

Intern Med. 2022 Apr 15;61(8):1247-1252. doi: 10.2169/internalmedicine.7526-21. Epub 2021 Oct 5.

Abstract

We herein report a case of recurrent multifocal, distal-dominant-sensorimotor neuropathy with ophthalmoplegia, IgM anti-GM1 antibody, and pyrexia-associated relapse. The patient developed sensory disturbance in her limbs after febrile disease at 50 years old. She had experienced several similar episodes and was admitted to the hospital at 56 years old. Based on a pathological study and electrophysiological findings consistent with chronic inflammatory demyelinating polyradiculoneuropathy (CIDP), maintenance IVIg therapy was administered and produced partial improvement with no relapse at one-year follow-up. Immunohistochemical studies suggested the presence of IgG (not IgM) anti-myelin antibodies. Chronic neuropathy with ophthalmoplegia and pyrexia-associated relapse may be a unique variant of CIDP.

Keywords: CIDP; fever; gangliosides; multifocal demyelinating neuropathy; ophthalmoplegia.

Publication types

  • Case Reports

MeSH terms

  • Chronic Disease
  • Female
  • Fever
  • Gangliosides
  • Humans
  • Immunoglobulin M
  • Ophthalmoplegia*
  • Phenotype
  • Polyradiculoneuropathy*
  • Polyradiculoneuropathy, Chronic Inflammatory Demyelinating* / complications
  • Polyradiculoneuropathy, Chronic Inflammatory Demyelinating* / diagnosis
  • Polyradiculoneuropathy, Chronic Inflammatory Demyelinating* / drug therapy
  • Recurrence

Substances

  • Gangliosides
  • Immunoglobulin M