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. 2024 Mar 19;16(4):805–822. doi: 10.1038/s44321-024-00053-x

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© The Author(s) 2024

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 licence, and indicate if changes were made. The images or other third party material in this article are included in the article’s Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article’s Creative Commons licence 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 licence, visit http://creativecommons.org/licenses/by/4.0/. Creative Commons Public Domain Dedication waiver http://creativecommons.org/publicdomain/zero/1.0/ applies to the data associated with this article, unless otherwise stated in a credit line to the data, but does not extend to the graphical or creative elements of illustrations, charts, or figures. This waiver removes legal barriers to the re-use and mining of research data. According to standard scholarly practice, it is recommended to provide appropriate citation and attribution whenever technically possible.

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Graph presenting retinal activity determined by ERG recordings at 3 mpi of Fam161a^tm1b/tm1b mice subretinally injected at PN14 with 10¹¹ GC of FCBR1-F0.4-HS (A), FCBR1-F0.4-HL (B) or FCBR1-F0.4-HS + FCBR1-F0.4-HL (C). FCBR1-F0.4 vectors improved the scotopic b-wave amplitudes to a different extent. Co-administration of both HS and HL isoforms enhances the best the b-wave amplitude compared to GFP-injected fellow eyes (two-way ANOVA, P = 0.0455), whereas HS or HL isoforms alone had a weak but significant increase in b-wave amplitudes (two-way ANOVA, P = 0.0101 and P = 0.0009, respectively, n = 5 eyes for all groups. Data are expressed as mean ± SEM). (D) Superposition of a representative scotopic single ERG response to 10 cd×s/m² of an HS + HL FAM161A-treated retina (violet) and its fellow eye treated with GFP (green). (E) Scotopic a-waves responses show a tendency to be improved by the co-treatment (P = 0.057). (F, G) The effect of co-administration of HS and HL isoform on the b-wave (P = 0.046 for the highest amplitudes) (F) and a-wave (G) amplitudes in photopic conditions is limited (n = 5 eyes for all groups. Data are expressed as mean ± SEM). Two-way ANOVA followed by Sidak’s multiple comparison test for comparison of the treated eyes to the fellow GFP-treated eyes for each stimuli intensity. *P < 0.05, **P < 0.01, ***P < 0.001, ****P < 0.0001. Data information: In total for four different experiments, 12, 10, and 11 eyes were injected for the HL, HS, and HS + HL groups, respectively. The five best ERG responses per group were then analyzed. Source data are available online for this figure.