Fig. 8

Loss of TMEM63A reduces MYO5A levels and disrupts Mbp mRNA transport in OLs (A) OL isolation workflow by MACS and subsequent bulk RNA-seq from mouse cortex. (B) Cell type-specific marker gene expression in T63a+/+ and T63a−/− cells at P11 and P35. (C and D) Differentially expressed genes (DEGs) between T63a+/+ and T63a−/− OLs at P11 (C) and P35 (D). An adjusted p value of <0.01 was considered significant. (E and F) Upregulated (E) and downregulated (F) biological processes associated with selected DEGs in T63a−/− OLs at P11. DEGs are grouped by biological processes. Node colors represent log2 fold expression changes in T63a−/− vs. T63a+/+ OLs. (G and H) Cortical OL at P11 immunostained for CNP (internodes, green) and MYO5A (red) in T63a+/+ (G) and T63a−/− (H) mice. Insets (dashed boxes) show internodes in higher magnification. (I and J) Mean MYO5A protein fluorescence intensity (I) and relative frequency distribution (with Gaussian fit, J) across CNP+ internodes. (K) Cortical T63a+/+ OL at P11, with Mbp mRNA (green) in the vicinity of MYO5A protein (red) in internodes marked with anti-CNP antibody (magenta). (L and M) Internodes marked with anti-CNP antibody (magenta) and Mbp mRNA (green). Insets (dashed boxes) show internodes in higher magnification. (N–P) Mean Mbp mRNA fluorescence intensity across CNP+ internodes (N) and relative frequency distribution (with Gaussian fit, P). Mbp mRNA raw integrated density values (Raw Int Den) vs. internode area (O). Slope, ∗p < 0.05 (P). Data represent mean ± SEM (I and N). ∗∗p < 0.01 (including P), ∗∗∗p < 0.001, ∗∗∗∗p < 0.0001 (including J). See also Figures S7 and S8 and Table S3 (sample sizes and statistical tests).