FIGURE

Fig. 4

ID

ZDB-FIG-240110-27

Publication

Accogli et al., 2024 - Variants in the WDR44 WD40-repeat domain cause a spectrum of ciliopathy by impairing ciliogenesis initiation

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Fig. 4

WDR44 variants affect zebrafish embryonic development.

a Dot blot analysis of WDR44 wild-type and variants expressed in zebrafish morphants (left). Protein loading was demonstrated by Ponceau staining of dot blot (right) probed with WDR44 antibodies. b Bright-field (BF) image analysis of zebrafish embryos injected with wdr44 morpholino and rescued with mRNA of human WDR44 wild-type or variants. Scale bar, 1 mm. (c–h) Quantification of developmental defects: body size, head dysmorphism, microphthalmia, body curvature, heart edema at 3 dpf, and distended pronephric tubules at 6 dpf. Statistical comparison with WT are shown. cP = 0.0229 (R733*), 0.0236 (G782C). dP = 0.0261 (L668S), 0.0062 (D669N), 0.0049 (R733*), 0.0052 (S764F), 0.0079 (G782C), 0.0355 (H839R). eP = 0.0157 (L668S), 0.0404 (D669N), 0.0099 (R733*), 0.0251 (S764F), 0.0046 (G782C), 0.0260 (H839R). fP = 0.0379 (D669N), 0.0032 (R733*), 0.0028 (S764F), 0.0204 (H839R). g 0.0204 (D669N), 0.0080 (R733*), 0.0067 (S764F), 0.0044 (G782C). hP = 0.0003 (D669N), 0.0053 (R733*), 0.0480 (G782C), 0.0331 (H839R), 0.0163 (N840S). i Representative images of 3 dpf Tg(wt1b:EGFP) embryos injected as in (b) showing developing pronephros. Scale bar, 50 μm. Mean ± s.e.m. from three independent experiments imaging >60 embryos (c–h) or >10 embryos (i). Unpaired two-tailed t-test; *P < 0.05, **P < 0.01, ***P < 0.001. Source data are provided as a Source Data file.

Expression Data

Expression Detail

Antibody Labeling

Phenotype Data

Phenotype Detail

Acknowledgments

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