Fig. 4

A truncation at the end of the motor domain of Dync2h1 leads to a cystic kidney phenotype. (A) Brightfield image of 5-dpf WT sibling and aa65.6^pd1086 mutant larvae. Red arrowhead indicates a kidney cyst. (B) Confocal images of transverse sections of 5-dpf WT sibling and aa65.6^pd1086 mutant larvae stained with phalloidin (red) and DAPI (blue). The arrowheads point to pronephric ducts and asterisks mark kidney cysts. (C) Confocal image of 3-dpf WT and aa65.6^pd1086 mutant embryos in whole-mount, stained for acetylated tubulin to mark cilia. (D) Linkage analysis for aa65.6^pd1086 using SNPtrack. (E) The homozygosity interval for aa65.6^pd1086. (F) Linkage analysis using single embryos. (G) Sequencing of genomic DNA of WT and aa65.6^pd1086 mutant larvae. (H) Injection of a morpholino directed against a splice site within the motor domain of dync2h1 phenocopies the aa65.6^pd1086 mutation. (I) Confocal images of transverse sections 3-dpf WT and dync2h1 morphants stained with phalloidin and DAPI, showing distended pronephric ducts (arrowheads). (J) Schematic of the Dync2h1 protein illustrating that the mutation removes a portion of the motor domain.