ZFIN Image: Chrystal et al., 2022, Fig. 2

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Figure Caption

Fig. 2

Zebrafish cfap20 mutants display developmental phenotypes characteristic of a motile ciliopathy.

a Schematic of CRISPR/Cas9 strategy targeting exon3. b The cfap20^ua5025 allele (cfap20⁻^/−) has a 1 bp deletion, 8 bp insertion (c.209delGinsTCGAGCTA; p.Gly70Valfs*29) that produces a frameshift at residue 70 and predicts loss of the majority of this deeply conserved protein (see also Supplementary Fig. ¹). c cfap20⁻^/− mutants have dramatically reduced abundance of transcript compared to wildtype at 48 h post fertilisation (hpf) (n = 4, error bars = SEM; two-tailed unpaired T-tests, gapdh P = 0.2912; cfap20 P = 1.54 * 10⁻⁷). d, e cfap20⁻^/− larvae exhibit anterior-posterior body axis kinks/ventral curvature (arrows) displayed at 48 hpf and 7 days post-fertilisation (dpf). e cfap20⁻^/− larvae develop pronephric duct cysts (arrowheads) at 7 dpf. f cfap20⁻^/− mutants exhibit significantly increased incidence of left-right pattern defects compared to wildtype (cardiac situs, n-value per condition shown, Fisher’s exact test, P = 0.0387). g, h cfap20⁻^/− adult homozygotes develop severe spine curvature compared to age-matched wildtype at 4 months post fertilisation. These motile ciliopathy phenotypes are also observed following knockdown of cfap20 in zebrafish (see Supplementary Figs. ³, ⁴). CRISPR clustered regularly interspaced short palindromic repeats, WT wildtype. Source data are provided as a Source Data file.

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Acknowledgments

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