Autoantibodies Against Trisulfated Heparin Disaccharide and Fibroblast Growth Factor Receptor-3 May Play a Role in the Pathogenesis of Neuropathic Corneal Pain

Cornea. 2023 Jul 1;42(7):821-828. doi: 10.1097/ICO.0000000000003142. Epub 2022 Oct 17.

Abstract

Purpose: The aim of this study was to describe cases of patients with presumable dysimmune small-fiber neuropathy (SFN)-related neuropathic corneal pain (NCP), presenting with autoantibodies against trisulfated heparin disaccharide (TS-HDS) or fibroblast growth factor receptor-3 (FGFR-3).

Methods: This study was a case series of 3 patients with NCP with positive anti-TS-HDS and/or anti-FGFR-3 autoantibodies and systemic SFN as confirmed by positive skin biopsy results.

Results: All 3 patients were women with a mean age of 34.3± 6.1 years. They suffered from moderate to severe persistent chronic ocular discomfort (10/10, 10/10, and 9/10 on a visual analogue scale, respectively). Although 1 patient suffered from ocular pain and photophobia alone, the other 2 patients experienced additional non-ocular pain. One of the patients had pain on her face and head, and 1 patient reported neck and lower back pain. Two patients had high anti-TS-HDS IgM titers, whereas 1 patient had both high anti-TS-HDS IgM and anti-FGFR-3 IgG titers. Skin biopsy confirmed the presence of SFN in all patients by demonstrating decreased intraepidermal nerve fiber density.

Conclusions: The presence of anti-TS-HDS and anti-FGFR-3 autoantibodies in patients with NCP with positive skin biopsy findings for SFN highlights the potential role of dysimmune SFN in the pathogenesis of this disease.

MeSH terms

  • Adult
  • Autoantibodies
  • Cornea / innervation
  • Female
  • Humans
  • Immunoglobulin M
  • Male
  • Neuralgia* / etiology
  • Receptors, Fibroblast Growth Factor*
  • Small Fiber Neuropathy* / etiology
  • Small Fiber Neuropathy* / pathology

Substances

  • Autoantibodies
  • FGFR3 protein, human
  • heparin disaccharide
  • Immunoglobulin M
  • Receptors, Fibroblast Growth Factor