March 2012
Volume 53, Issue 14
Free
ARVO Annual Meeting Abstract  |   March 2012
Fundus Autofluorescence in the Abca4-/- Mouse, an Animal model for Stargardt disease - Correlation with Retinal Function, Histology and Accumulation of A2E
Author Affiliations & Notes
  • Peter Charbel Issa
    Nuffield Laboratory of Ophthalmology, University of Oxford, Oxford, United Kingdom
  • Alun R. Barnard
    Nuffield Laboratory of Ophthalmology, University of Oxford, Oxford, United Kingdom
  • Mandeep S. Singh
    Nuffield Laboratory of Ophthalmology, University of Oxford, Oxford, United Kingdom
  • Emma Carter
    Nuffield Laboratory of Ophthalmology, University of Oxford, Oxford, United Kingdom
  • Roxana A. Radu
    Ophthalmology, UCLA/Jules Stein Eye Inst, Los Angeles, California
  • Ulrich Schraermeyer
    Experimental Vitreoretinal Surgery, Institute for Ophthalmic Research, Tubingen, Germany
  • Robert E. MacLaren
    Nuffield Laboratory of Ophthalmology, University of Oxford, Oxford, United Kingdom
  • Footnotes
    Commercial Relationships  Peter Charbel Issa, None; Alun R. Barnard, None; Mandeep S. Singh, None; Emma Carter, None; Roxana A. Radu, None; Ulrich Schraermeyer, None; Robert E. MacLaren, None
  • Footnotes
    Support  European Commission, FP7, Marie Curie Intra-European Fellowship 237238; Fight for Sight, Wellcome trust (086868/Z/08/Z); Oxford Stem Cell Institute; Health Foundation
Investigative Ophthalmology & Visual Science March 2012, Vol.53, 4988. doi:
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      Peter Charbel Issa, Alun R. Barnard, Mandeep S. Singh, Emma Carter, Roxana A. Radu, Ulrich Schraermeyer, Robert E. MacLaren; Fundus Autofluorescence in the Abca4-/- Mouse, an Animal model for Stargardt disease - Correlation with Retinal Function, Histology and Accumulation of A2E. Invest. Ophthalmol. Vis. Sci. 2012;53(14):4988.

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      © ARVO (1962-2015); The Authors (2016-present)

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Abstract

Purpose: : To investigate the feasibility of quantitative and qualitative fundus autofluorescence (AF) assessment for monitoring lipofuscin accumulation in the Abca4-/- mouse, and to correlate findings with retinal function, histology and measurements of A2E.

Methods: : Confocal scanning laser ophthalmoscopy (cSLO) was used for in vivo fundus AF excited at 488 nm in pigmented Abca4-/- mice and wild type controls. For quantitative AF analysis, the mean grey level on fundus AF images acquired with standardized detector sensitivity was measured within a ring shaped midperipheral area. Additional analysis included electroretinography (ERG), post-mortem histological assessment of the outer nuclear layer thickness, and measurements of A2E using HPLC.

Results: : Fundus AF intensity was similar in four weeks old animals. With age, a steady increase of AF occurred, which was more pronounced in Abca4-/- mice compared to wild type controls. Fundus AF was significantly higher (p<0.01) in Abca4-/- mice at 9 weeks and older. The grey level ratio between Abca4-/- and age-matched wild type mice, which accounts for confounding factors such as pupil size or other age-related changes, was 1.2 at 4 weeks, 2.0 at 3 months, and 2.25 at 6 months. A fleck pattern of high fundus AF was clearly visible in 6 month old Abca4-/- mice, but not in wild type controls. In Abca4-/- mice 12 months and older, there were spots of decreased AF suggesting atrophic changes of the retinal pigment epithelium.ERG a-wave and b-wave amplitudes and the outer nuclear layer thickness were not significantly different between Abca4-/- mice and wild type controls up to an age of 9 months. Total A2E measurements increased similar to fundus AF measures. However, the ratio was 4-5 times higher than on fundus AF measures, possibly due to other fluorophores contributing to the overall difference.

Conclusions: : cSLO imaging in mice allows detecting significant differences in fundus AF intensity and pattern between Abca4-/- mice and wild type controls in age groups that show no significant difference in photoreceptor function (ERG) and number (outer nuclear layer thickness). Thus, fundus AF imaging is a useful technique to assess potential treatments aiming at reducing lipofuscin accumulation in the retinal pigment epithelium.

Keywords: imaging methods (CT, FA, ICG, MRI, OCT, RTA, SLO, ultrasound) • retinal pigment epithelium 
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