Double hyperautofluorescent ring on fundus autofluorescence in ABCA4

Ophthalmic Genet. 2018 Jan-Feb;39(1):87-91. doi: 10.1080/13816810.2017.1335330. Epub 2017 Jul 20.

Abstract

We report an unusual phenotype in a child with a clinical diagnosis of recessive Stargardt disease (STGD1) and two pathogenic variants in the ABCA4 gene. Typically, the diagnosis of early-onset STGD1 is challenging because children may present with a variety of fundus changes and a variable rate of progression. At the time of his initial visit, the 6-year-old boy presented with 20/200 OD (right eye) and 20/150 OS (left eye), symmetrical mild foveal atrophy without flecks on fundus exam, and foveal hypoautofluorescence surrounded by a homogeneous hyperautofluorescent background on wide-field fundus autofluorescence. Over 4 years of follow-up, the retinal atrophy continued to progress, resulting in two well-defined and concentric hyperautofluorescent rings: one ring located at the posterior pole and the other located around the peripapillary region. Visual acuity also deteriorated to counting fingers at 4ft OD and 20/500 OS. To the best of our knowledge, this phenotype has not been previously described with the ABCA4 gene.

Keywords: ABCA4; Stargardt disease; fundus autofluorescence; macular degeneration; retinal degeneration; retinal imaging.

Publication types

  • Case Reports

MeSH terms

  • ATP-Binding Cassette Transporters / genetics*
  • Child
  • Fluorescein Angiography
  • Fundus Oculi
  • Genetic Variation*
  • Humans
  • Macular Degeneration / congenital*
  • Macular Degeneration / diagnosis
  • Macular Degeneration / genetics
  • Macular Degeneration / physiopathology
  • Male
  • Optical Imaging
  • Phenotype
  • Retina / pathology*
  • Retrospective Studies
  • Stargardt Disease
  • Tomography, Optical Coherence
  • Vision Disorders / genetics
  • Vision Disorders / physiopathology
  • Visual Acuity / physiology
  • Visual Fields / physiology

Substances

  • ABCA4 protein, human
  • ATP-Binding Cassette Transporters