IN VIVO OBSERVATION OF RETINAL VASCULAR DEPOSITS USING ADAPTIVE OPTICS IMAGING IN FABRY DISEASE

Retina. 2020 Aug;40(8):1623-1629. doi: 10.1097/IAE.0000000000002648.

Abstract

Purpose: To report a novel finding in patients with Fabry disease, that is, the observation by adaptive optics ophthalmoscopy of intracellular lipidic deposits in retinal vessels.

Methods: Observational two-center case series. Eighteen patients with genetically proven Fabry disease underwent flood-illumination adaptive optics ophthalmoscopy imaging (rtx1; Imagine Eyes, Orsay, France) of retinal vessels.

Results: Fourteen patients (78% of all patients; 7 of the 10 women and 7 of the 8 men) showed paravascular punctuate or linear opacities in both eyes. In the least-affected patients, these were seen only in the wall of precapillary arterioles as discrete spots of 5 µm to 10 µm large, whereas in those more severely affected, capillaries and first-order vessels were also involved with diffuse opacification of the wall. These deposits sometime showed a striated pattern, suggesting colocalization with vascular smooth muscle cells.

Conclusion: Adaptive optics ophthalmoscopy of retinal vessels may be of interest for patients with Fabry disease, providing noninvasive, gradable evaluation of microvascular involvement.

Publication types

  • Multicenter Study

MeSH terms

  • Adult
  • Aged
  • Arterioles / diagnostic imaging
  • Arterioles / pathology*
  • Fabry Disease / diagnostic imaging
  • Fabry Disease / physiopathology*
  • Female
  • Humans
  • Male
  • Middle Aged
  • Muscle, Smooth, Vascular / diagnostic imaging
  • Muscle, Smooth, Vascular / metabolism*
  • Ophthalmoscopy / methods*
  • Optics and Photonics
  • Retinal Artery / diagnostic imaging
  • Retinal Artery / pathology*
  • Sphingolipids / metabolism*
  • Young Adult

Substances

  • Sphingolipids