FIGURE

Fig. 2

ID
ZDB-FIG-221109-11
Publication
Chrystal et al., 2022 - The inner junction protein CFAP20 functions in motile and non-motile cilia and is critical for vision
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Fig. 2

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

a Schematic of CRISPR/Cas9 strategy targeting exon3. b The cfap20ua5025 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.

Expression Data
Gene:
Fish:
Anatomical Term:
Stage: Long-pec

Expression Detail
Antibody Labeling
Phenotype Data
Fish:
Observed In:
Stage Range: Long-pec to Adult

Phenotype Detail
Acknowledgments
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