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. 2021 Mar 2;15:939–952. doi: 10.2147/OPTH.S292527

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© 2021 Aleman et al.

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Changes in retinal sensitivity and performance of RPE65-LCA patients on the virtual reality orientation and mobility test after gene therapy.Notes: (A) NIR-FAF, 55°-wide images of the posterior retina of the right eye of the two with RPE65-LCA treated with bilateral subretinal gene therapy (Luxturna, Sparks Therapeutics Inc., Philadelphia, PA, USA). Red line denotes the inferior boundary of a subretinal bleb that contains the treating agent, which extended from the superior retina crossing the fovea and into the inferior pericentral retina. (B and C) Changes in retinal sensitivity measured by automated static perimetry (B) and FST (C) after gene therapy. (B) Light-adapted achromatic and dark-adapted two-color chromatic static perimetry (shown only responses to a blue 500 nm stimulus) in the patients before (dashed lines) and after (continuous line) gene therapy. Dotted lines define lower limit (mean – 2SD) of sensitivity in control subjects. S, superior; I, inferior visual field. Horizontal arrows show the improvement in sensitivity supporting a treatment effect. (C) FST sensitivity estimates measured with spectral stimuli (blue, 467 nm; red, 637 nm) in dark-adapted (>30 min) patients. Dotted gray line is the lower limit (mean-2SD) of the sensitivity to the short wavelength 467 nm stimulus in control subjects. Values are converted into positive dB values from possible negative outputs from the FST instrument. (D) Summary VR-O&M parameters in patients compared to control subjects in this study for timing orientation parameter in the “arrows only” test (left panel), and for collisions (middle panel) and performance timing (right panel) of the “arrows plus obstacles” test. Shown are mean values + 2SD. Data points for patients before treatment (pre-Tx = black symbols) are connected to post-treatment (post-Tx; white symbols) values to demonstrate the main shift in performance. VR21 did not undergo “arrows only” testing post-treatment. Panels A-C reproduced with permission from Maguire AM, Bennett, J, Aleman E, et al. Clinical Perspective: Treating RPE65-Associated Retinal Dystrophy.Mol Ther. 2021;29(2):442–463.⁵