Investigation of ocular involvement in patients with Fabry disease

Ann Med. 2023 Dec;55(1):2226909. doi: 10.1080/07853890.2023.2226909.

Abstract

Purpose: To investigate ocular abnormalities in Fabry disease (FD).

Methods: Forty-five patients with FD diagnosed by genetic analysis were enrolled in a single medical centre. The following ocular examinations were performed: slit-lamp examination, ophthalmic fundus imaging, in vivo confocal microscopy (IVCM) and optical coherence tomography (OCT). The prevalences of typical abnormalities in the cornea, conjunctiva and retina were recorded; their differences between hemizygote and heterozygote were compared.

Results: In this study, the prevalence of corneal verticillata was 97.8% (44/45). Corneal examination with IVCM demonstrated hyper-reflective intracellular inclusions located within basal epithelial cells. Conjunctival vessel malformations were observed in 64.4% (29/45) of patients, and retinal vessel tortuosity was observed in 62.2% (28/45) of patients. OCT revealed many strong hyper-reflective foci (HRF) in the inner retinal layer (in 66.7% [30/45] of patients); these foci may represent retinal vascular plexi. The prevalences of conjunctival vessel malformation, retinal vessel tortuosity and HRF were higher in hemizygote than in heterozygote.

Conclusions: Corneal verticillata, HRF on OCT, conjunctival vessel malformation and retinal vessel tortuosity exhibit high prevalence in patients with FD. These ocular manifestations are characteristic and easily accessible; thus, they should be considered diagnostic criteria for FD.

Keywords: Fabry disease; corneal verticillata; optical coherence tomography; retinal vessel tortuosity.

Plain language summary

The main ocular features of the patients with FD are corneal verticillata, HRF on OCT, conjunctival vessel malformation and retinal vessel tortuosity.These ocular manifestations should be considered as the diagnostic criteria of FD.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Conjunctiva
  • Cornea / diagnostic imaging
  • Eye Diseases*
  • Fabry Disease* / complications
  • Fabry Disease* / diagnosis
  • Fabry Disease* / genetics
  • Humans
  • Tomography, Optical Coherence

Grants and funding

This work was supported by ULM-PUHSC Joint Institute for Translational and Clinical Research (No: PKU2017ZC001-5) and the National Natural Science Foundation (China) (No: 81100840).