Fig 1: Overexpression of human wild-type DCTN1 rescues the CaP growth defects at 6dpf and the behavioral deficits at 2dpf. a CaP morphological defects seen at 6dpf in mutant larvae are rescued by single-cell overexpression of DCTN1-GFP. b Quantification of cell tracings show recovery of arbors size in rescued mutant larvae, as determined by total projection number and total cell length, without affecting average projection length. Overgrowth is due to lack of competition by neighboring mutant cells. c DCTN1-GFP is found to accumulate at synaptic sites (close-up of heatmap), d as confirmed by colocalization (in white) in double immunohistochemistry of DCTN1-GFP (anti-GFP in cyan) with post-synaptic ACh receptors (a-bungarotoxin, in magenta). d Overexpression of exogenous DCTN1-GFP by injection of 400 ng/ul RNA rescued the NMJ functional defects leading to impaired locomotor behavior in 2dpf embryos. Touch-evoked escape response was quantified and is shown as ratios relative to the average values obtained for wild-type escapes for duration, distance, and maximum instant speed. Data shown as c) average +/- SD, d) median +/- interquartile range (b: n cells = 19, 13; d:n embryos = 26, 24, 19). Scale bar = 50 µm