(A) mbp:gfp expression in control and (B) 25 μM forskolin (FSK) pulsed gpr126st63 5 dpf larvae. Brackets denote spinal cord, arrows indicate PLLn, dotted lines indicate melanocytes obscuring PLLn. (C) Quantification of mbp:gfp as a percentage of larvae with each mbp PLLn phenotype at 5-6 dpf. *** p<0.001, Fisher’s Exact Test, “non-st63-like” vs “st63-like.” (D) Transformation of data from panel C. Dots indicate individual larva and are jittered to show all samples. Bars indicate mean ± SD. *** p<0.001, Student’s t-test. (E) Quantification of mbp:gfp(+) motor axons at 5-6 dpf. NS = no significant difference. (F) PLLn score and mbp:gfp(+) motor axons for 1462 Pharmakon compounds. Each square = one compound. Dots are jittered and transparent to show all samples. (G) Workflow of screens for gpr126st63 suppressor compounds. (H) Rolipram restores PLLn mbp:gfp but not (I) motor axons in gpr126st63. * p<0.05, Fisher’s Exact Test, “non-st63-like” vs “st63-like”.