Fig. 2

The vu76 allele is a mutation of dync1h1. A–D: Compound heterozygous vu76/dync1h1^hi3684Tg, homozygous dync1h1^hi3684Tg and dync1h1 MO-injected larvae have similar phenotypic characteristics including small heads and small eyes (e) and abnormally dispersed pigment (arrows). E: Western blotting of protein extracts obtained from 3 and 5 dpf larvae with anti-Dync1h1 antibody reveals an approximately 500 kD protein present in 3 and 5 dpf wild-type larvae and 3 dpf mutant larvae but absent from 5 dpf mutant larvae (arrow). The blot was probed with anti-α-Tubulin antibody as a loading control. F: Sequencing of dync1h1 cDNA from wild-type and vu76 mutant larvae revealed a G to A transition changing a tryptophan codon to a stop codon. This change is predicted to prematurely terminate translation at amino acid position 3561, within the motor domain of Dync1h1 (G). H–M: Whereas 3 dpf wild-type larvae rapidly aggregate melanosomes into tight spots upon treatment with epinephrine, which reflects retrograde transport of melanosomes on microtubules, melanosome distribution in vu76 mutant larvae is not changed by epinephrine. Images in H–M show dorsal views of the head. Elapsed time following initiation of epinephrine treatment is indicated above the panels.