Fig. 2

Fig. 2

Isolation of new mutant alleles for ift172 and lrrc6 that cause kidney cyst formation. (A) Brightfield image of 5-dpf WT sibling and ccn1.9^pd1085 homozygous mutant. Red arrowhead indicates a kidney cyst. (B) Confocal images of transverse sections of 5-dpf WT sibling and ccn1.9^pd1085 mutant stained with phalloidin (red) and DAPI (blue). Arrowheads point to the pronephric ducts and asterisks mark kidney cysts. (C) Linkage analysis for ccn1.9^pd1085 using SNPtrack. (D) The homozygosity interval for ccn1.9^pd1085. (E) Sequencing of genomic DNA of WT and ccn1.9^pd1085 mutant larvae. Mutants bear a G-to-A mutation at an essential splice site in exon 13 of ift172 (underlined). (F) Brightfield image of 5-dpf WT sibling, ift172^{hi2211Tg/pd1085} and ift172^hi2211Tg larvae. (G) Brightfield image of 5-dpf WT sibling and dx33.9^pd1087 mutant larvae. (H) Confocal images of transverse sections of 5-dpf WT sibling and dx33.9^pd1087 mutant larvae stained with phalloidin and DAPI as in B. (I) Linkage analysis for dx33.9^pd1087 using SNPtrack. (J) The homozygosity interval for dx33.9^pd1087. (K) Sequencing of genomic DNA of WT and dx33.9^pd1087 mutant larvae. Mutants are homozygous for a T-to-G nonsense mutation. (L) Rescue of the dx33.9^pd1087 phenotype via injection of WT lrrc6 cRNA.