Fig. 1

The Tg(ubi:loxP‐ift54‐loxP‐myr‐mcherry,myl7:EGFP)sh488 rescue transgene provides partial rescue of homozygous ift54 ^tp49 mutants. A: Diagram of the ubi:loxP‐ift54‐loxP‐myr‐mcherry,myl7:EGFP transgene. B: Five‐dpf siblings from an ift54 ^tp49/+;Tg(ubi:loxP‐ift54‐loxP‐myr‐mcherry,myl7:EGFP)sh488 x ift54 ^tp49/+ cross. Sequencing traces show the ift54 ^tp49 C‐to‐T mutation genotype (asterisk denotes stop codon). Homozygous mutant ift54 ^tp49 (ift54−/−) embryos without the transgene display the expected curly‐down phenotype. The Tg(ubi:loxP‐ift54‐loxP‐mcherry,myl7:EGFP)sh488 transgene (ubi:ift54) does not affect normal development compared with WT but rescues the phenotype of some ift54 ^tp49 homozygotes. C: Proportion of phenotypes of the transgenic (+) and nontransgenic (−) embryos from six ift54^tp49/+;Tg(ubi:loxP‐ift54‐loxP‐myr‐mcherry,myl7:EGFP)sh488 ^/+ x ift54 ^tp49/+ crosses. Total number of embryos is shown above each column. The extent of rescue varied considerably between clutches. D: Five‐month‐old adult fish alongside sequencing traces showing the ift54 ^tp49 C‐to‐T mutation genotype. ift54 ^tp49 mutants with the Tg(ubi:loxP‐ift54‐loxP‐myr‐mcherry,myl7:EGFP)sh488 transgene (ift54‐;ubi:ift54) can survive into adulthood but develop scoliosis. Dpf, days postfertilization; WT, wild‐type