Azi1 (Cep131) knockdown causes defects in KV formation, melanosome trafficking and vision defects in zebrafish.

Embryos were untreated or injected with 125 µM Bbs4 MO or 150 µM cep131 MO as previously described [28], [29], [31], [33]. A) azi1, as well as bbs4 morpholino injection causes a mild to severe body curvature phenotype in the 48 hpf larva. Images indicate examples of normal, mild or severe phenotypes. B) Quantification of body curvature. More of the azi1 morphants have severe body curvature than bbs4 morphants. C) Micrographs show KVs from MO injected and uninjected 8–10 somite stage embryos. D) bbs4 MO caused KV abnormalities in 30% of embryos, and azi1 MO caused 35% abnormal KV compared with 12% in control embryos (P<0.001 Fisher's exact test). E) bbs4 MO and azi1 MO delayed melanosome retrograde transport to 4.04+/−0.43 min, and 3.03+/−0.12 min, respectively compared to the control MO, in which the complete transport occurs within 1.92+/−0.04 minutes. F) Vision assays were performed on the morphants by observing their response to dark/light cues as previously described [33], [35]. For each fish the vision response tested 5 times. For C and D data are presented mean +/− SEM, P<0.01 by one-way ANOVA with Tukey post-test. The number of embryos used is presented in the figure.