Figure 2. Suppression of usp38 (USP35 ortholog in zebrafish) ameliorates the defects caused by depletion of bbs4.

(A) Morpholinos were injected as described in the Methods. Depletion of bbs4 results in CE defects, including wider anterior-posterior body gap, somite, and loss of eyes (mostly in Class II). To quantitatively determine the CE phenotype, the angle of body gap was measured in each embryo, and the embryos were classified as described in Methods. Coinjection of usp38-MO reduces both Class I and Class II embryos. Arrowheads represent the tip of anterior and posterior body axis. Brackets represent the width of somite. (χ² analysis, *P < 0.05, ****P < 0.0001). (B) Depletion of bbs4 leads to the atrophy and deficient convolution in the proximal tubules, a phenotype that is related to cyst formation. Knockdown of usp38 expression ameliorates renal defects seen in bbs4 morphants (χ² analysis, ***P < 0.001). Scale bar: 100 μm. (C) While bbs4 CRISPR (mosaic) mutant exhibit defects in the convolution of proximal tubules, deletion of usp38 ameliorates these defects (χ² analysis, **P < 0.01, ****P < 0.0001).