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[Preprint]. 2025 Nov 5:2025.11.03.686369. [Version 1] doi: 10.1101/2025.11.03.686369

Figure 1. Specc1l mutants have shortened primary cilia in vitro and in vivo.

Figure 1.

A-C. Embryonic day (E) 13.5 mouse palate mesenchyme tissue from wildtype (WT) (A), Specc1lΔEx4/ΔEx4 null (B), and Specc1lΔCCD2/ΔCCD2 (C) embryos, stained with acetylated-α-tubulin (green), ARL13B (red), and DAPI. The insets show 2x zoom of select regions. D. Quantification of cilia length from 3 different embryos of each genotype (WT, n=250; ΔEx4, n=290; ΔCCD2, n=200, **** <0.0001, Scale bar represents 2μm). E-G. Mouse embryonic fibroblasts (MEFs) from WT (E), Specc1lΔEx4/ΔEx4 (F), and Specc1lΔCCD2/ΔCCD2 (G) embryos stained with acetylated-α-tubulin (green), F-actin (red), and DAPI. H. Quantification of cilia length from 3 different experiments (n=150 cilia/genotype, **** <0.0001, Scale bar represents 2μm). I-L. MEFs from WT (I), Specc1lΔEx4/ΔEx4 (J), and Specc1lΔCCD2/ΔCCD2 (K) stained with acetylated-α-tubulin (green) and imaged at 100x with 4x zoom revealed bulbous tips in Specc1lΔCCD2/ΔCCD2 (K). L. Quantification of bulbous tips from 3 different experiments (n=150 cilia/genotype, *** <0.001, Scale bar represents 2μm). M-O. E13.5 mouse palate mesenchyme tissue from WT (M), Specc1lΔEx4/ΔEx4 (N), and Specc1lΔCCD2/ΔCCD2 (O) stained with GLI1 (white). P. Quantification of GLI1 from 3 different embryos of each genotype (** <0.0044, **** <0.0001, Scale bar represents 2μm, Error bars represent 95% CI for (D &H) and Mean ± standard deviation (SD) for (L &P)).