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. 2020 Oct 19;29(20):3373–3387. doi: 10.1093/hmg/ddaa228

Figure 6.

Figure 6

Rx3-Cre-mediated disruption of Nf2 results in defective alignment of OF margins during the closure process. (A–D) F-actin localization in the OF at E11.0 (A, B) and at E11.5 (C, D) in control (A, C) and Nf2CKO optic cups (B, D). A: In E11.0 control embryos, the OF margins become tightly aligned, shaped with a convex surface toward the basal space (arrowheads). (B) In the Nf2CKO OF, shape of the basal surface of the OF margins is convex, similar to controls, however, alignment of the margins is less advanced (B, arrowheads). At E11.5, the OF margins in controls are well aligned along most of the OF with flattened basal surfaces (C, arrowheads). The RPE layer consists of cuboidal cells (bracket). (D) In Nf2CKO eyes, the OF margins align only partially (arrowheads), exhibit convex shape, and OF cells continuous with the RPE layer show increased height in the apicobasal axis, thereby appearing columnar (bracket). (E) Quantification of aligned OF margins. Extent of attached margins was measured in the OF, as outlined in representative examples shown in C, D for aligned OF between arrowheads. In Nf2CKO, few margins show close alignment, which is often much reduced, compared with controls (lack of alignment defined = 0). Data are means ± SD and Student’s T-test was applied for statistical analysis. (F) Analysis of cellular height along the apicobasal axis of cells lining the ventral OF margins (see brackets in C, D; Supplementary Material, Figure S3G) reveals significantly increased cellular height in the temporal OF margin. Statistical analysis was performed using nested T-test, data are means (with minimal and maximal points). P-values are indicated on the horizontal lines in each graph , significance < 0.05 (E, F). (G) Proposed model summarizing our observations, for explanation, see text (left: control optic cup; right: Nf2CKO). The arrows point to potential effects of RPE hyperproliferation on the mechanodynamics of optic cup growth and apposition of OF margins. Sagittal view with nasal orientation to the left. (NR: neural retina, L: lens, N: nasal, T: temporal). Scale bars: 50 μm in A, C.