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. 2017 Nov 23;7:16151. doi: 10.1038/s41598-017-16275-8

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© The Author(s) 2017

Open Access This article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The images or other third party material in this article are included in the article’s Creative Commons license, unless indicated otherwise in a credit line to the material. If material is not included in the article’s Creative Commons license and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this license, visit http://creativecommons.org/licenses/by/4.0/.

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Retinal function and axial length recovery after AAV2/8-mMfrp transduction. (A) Fundus autofluorescence (AF) of Mfrp ^rd6 /Mfrp ^rd6 versus AAV2/8-mMfrp treated eye. A fluorescence standard (an intensity comparison) appears as a bright strip at the top of each image. The AAV2/8-mMfrp eye fluoresced with reduced AF intensity. (B) SD-OCT suggests rescue of retinal cell layers. Representative SD-OCT image shows cell layer thickness was 230 µm in the untreated eye and 242 µm in the treated eye. (C) Quantification of ERG b-wave amplitude shows a significant increase in retinal bipolar cell activity 2-months following gene therapy. Data were analyzed with a pairwise Student’s t-test (p = 0.04). (D) Quantification of ERG a-wave amplitude showing significant increase in photoreceptor cell function 2-months following gene therapy (p = 0.004). (E) Eye axial length was measured using the 50 MHz ultrasound bio-microscope (UBM) probe on an AVISO A/B (Quantel Medical) and reported in millimeters (8 eyes per group). Data were analyzed using 1-way ANOVA followed by Tukey’s multiple comparison test. AAV2/8-mMfrp mice had a significant increase (average of 0.1 mm) in axial length compared to Mfrp ^rd6 /Mfrp ^rd6 mice (p = 0.0334). Histological comparison of Mfrp ^rd6 /Mfrp ^rd6 (F) and AAV2/8-mMfrp mice (G) eye axial length.