Jobst-Schwan et al., 2018 - Acute multi-sgRNA knockdown of KEOPS complex genes reproduces the microcephaly phenotype of the stable knockout zebrafish model. PLoS One   13:e0191503 Full text @ PLoS One

Fig. 4 ZFIN is incorporating published figure images and captions as part of an ongoing project. Figures from some publications have not yet been curated, or are not available for display because of copyright restrictions.

PHENOTYPE:
Fish:
Observed In:
Stage Range: Protruding-mouth to Day 5

Fig. 5 Acute multi-sgRNA CRISPR/Cas9 knockout reproduces the microcephaly phenotype of stable osgep and tprkb KO lines. Larvae were imaged from a dorsal view. Head diameter to total body length was calculated. This ratio was normalized by the mean of the uninjected/wildtype control and defined as “microcephaly index” (MI). MI was determined at 6 dpf except for the stable KO line osgep c.102_105del at 4 dpf due to early lethality (see Fig 4a). (a) Representative phenotypes of a larva that was injected with the osgep targeting multi-sgRNA pool compared to a scrambled control larva. The red lines display the typical axes used for the measurements. (b) For osgep, the acute multi-sgRNA KO showed a significant microcephaly compared to uninjected and scrambled control. (c) The microcephaly phenotype is recapitulated in the homozygous larvae of the stable KO line osgep c.102_105del compared to wildtype and heterozygous clutch mates. (d) For osgep, MI is significantly higher in acute KO larvae compared to homozygous stable KO larvae. (e) For tprkb, the acute multi-sgRNA KO showed a significant microcephaly compared to uninjected and scrambled control. (f) The microcephaly is recapitulated in the homozygous larvae of the stable KO line tprkb c.370_376delinsAA compared to wildtype and heterozygous clutch mates. (g) For tprkb, no significant difference in MI was found for acute KO larvae compared to homozygous stable KO larvae

PHENOTYPE:
Fish:
Observed In:
Stage Range: Day 4 to Day 5
Acknowledgments:
ZFIN wishes to thank the journal PLoS One for permission to reproduce figures from this article. Please note that this material may be protected by copyright. Full text @ PLoS One