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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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(A) Quantification of ONL thickness throughout the whole retina revealed that the combination of HS + HL (n = 5) was the most efficient to rescue the retina in comparison to the GFP group (P varies from <0.05 to 0.0001) or the HL group (n = 5). The mean (gray line) and SD (dotted gray lines) of the ONL thickness in the WT retina are indicated as reference. A two-way ANOVA followed by a Tukey’s test to determine significant differences at specific locations between the FAM161A and GFP-treated eyes was performed. (B) The ONL thickness rescue by FCBR1-F0.4-FAM161A treatment was dependent on the density of FAM161A-positive CC. The highest FAM161A-positive CC density (nb of CC/200 μm) and the thicker ONL rescue were observed in the retina treated with the combination of FCBR1-F0.4-HS + FCBR1-F0.4-HL. One-way ANOVA followed by a Tukey’s test for multiple comparisons of all the groups. n = 5 eyes per group, and 13–22 regions of 200 µm per eye were analyzed. (C) The relation between CC density and ONL thickness can be described by a linear regression (plain line). The slope of FCBR1-F0.4-HL + HS linear regression (slope = 0.5267; R² = 0.4467) is higher than the slope of FCBR1-F0.4-HL (slope = 0.1885; R² = 0.2147) or FCBR1-F0.4-HS (slope = 0.3224; R² = 0.5925) (P < 0.01). KO Fam161a^tm1b/tm1b, WT wild type, ONL outer nuclear layer, CC connecting cilium. Data are expressed as mean ± SEM. *P < 0.05, **P < 0.01, ***P < 0.001, ****P < 0.0001. Source data are available online for this figure.