Quantification of peripapillary sparing and macular involvement in Stargardt disease (STGD1)

Invest Ophthalmol Vis Sci. 2011 Oct 10;52(11):8006-15. doi: 10.1167/iovs.11-7693.

Abstract

Purpose: To quantify and compare structure and function across the macula and peripapillary area in Stargardt disease (STGD1).

Methods: Twenty-seven patients (27 eyes) and 12 age-similar controls (12 eyes) were studied. Patients were classified on the basis of full-field electroretinogram (ERG) results: Fundus autofluorescence (FAF) and spectral domain-optical coherence tomography (SD-OCT) horizontal line scans were obtained through the fovea and peripapillary area. The thicknesses of the outer nuclear layer plus outer plexiform layer (ONL+), outer segment (OS), and retinal pigment epithelium (RPE) were measured through the fovea, and peripapillary areas from 1° to 4° temporal to the optic disc edge using a computer-aided, manual segmentation technique. Visual sensitivities in the central 10° were assessed using microperimetry and related to retinal layer thicknesses.

Results: Compared to the central macula, the differences between controls and patients in ONL+, OS, and RPE layer thicknesses were less in the nasal and temporal macula. Relative sparing of the ONL+ and/or OS layers was detected in the nasal (i.e., peripapillary) macula in 8 of 13 patients with extramacular disease on FAF; relative functional sparing was also detected in this subgroup. All 14 patients with disease confined to the central macula, as detected on FAF, showed ONL+ and OS layer thinning in regions of normal RPE thickness.

Conclusions: Relative peripapillary sparing was detected in STGD1 patients with extramacular disease on FAF. Photoreceptor thinning may precede RPE degeneration in STGD1.

Publication types

  • Research Support, N.I.H., Extramural
  • Research Support, Non-U.S. Gov't
  • Research Support, U.S. Gov't, Non-P.H.S.

MeSH terms

  • ATP-Binding Cassette Transporters / genetics
  • Adolescent
  • Adult
  • Electroretinography
  • Female
  • Fluorescein Angiography
  • Genotype
  • Humans
  • Macular Degeneration / congenital
  • Macular Degeneration / diagnosis*
  • Macular Degeneration / genetics
  • Male
  • Middle Aged
  • Optic Disk / pathology*
  • Prospective Studies
  • Retinal Photoreceptor Cell Inner Segment / pathology*
  • Retinal Pigment Epithelium / pathology*
  • Rod Cell Outer Segment / pathology*
  • Stargardt Disease
  • Tomography, Optical Coherence
  • Visual Acuity / physiology
  • Visual Field Tests
  • Visual Fields
  • Young Adult

Substances

  • ABCA4 protein, human
  • ATP-Binding Cassette Transporters