FIGURE

Fig. 7.

ID

ZDB-FIG-210814-59

Publication

Cardenas-Rodriguez et al., 2021 - Genetic compensation for cilia defects in cep290/NPHP6 mutants by upregulation of cilia-associated small GTPases

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Fig. 7.

Upregulation of small GTPases/chaperones that mediate cilia lipidated protein delivery rescue cep290ex25 morphant axoneme defects. (A) Overexpression of cilia GTPases/chaperone (arl3, arl13b and unc119b) in cep290ex25 morphant embryos partially rescued the curly axis cilia defect related phenotype. (B) arl13b overexpression was sufficient to rescue Kupffer's vesicle (KV) cilia length defects observed in stage-matched cep290ex25 morphant embryos. (C) Kupffer's vesicle cilia length was also rescued by overexpression of unc119b mRNA. (D) Injection of arl3 mRNA only partially rescued Kupffer's vesicle cilia length in cep290ex25 morphant embryos. (E) The injection of arl3, arl13b and unc119b mRNAs also was sufficient to rescue the photoreceptor outer segment length defect on cep290ex25 morphants. *P<0.01; ***P<0.001; ****P<0.0001; ns, not significant (Dunnett's or Tukey's test). y-axis values represent the average length of >70 Kupffer's vesicle cilia from 3-6 embryos per condition or of >200 OS from four embryos per condition. Data are mean±s.d.

Expression Data

Expression Detail

Antibody Labeling

Phenotype Data

Fish:	AB/TU + MO7-cep290
Knockdown Reagent:	MO7-cep290
Observed In:	axis Kupffer's vesicle cilium photoreceptor outer segment layer post-vent region
Stage Range:	10-13 somites to Protruding-mouth

Phenotype Detail

Acknowledgments

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