Figure 1

rpgrip1l-/- juvenile fish develop scoliosis, which is rescued by RPGRIP1L expression in foxj1a-positive cells. (A) Representative rpgrip1l-/- fish at 5 days post-fertilization (dpf) (4 mm body length, larvae), 5 weeks pf (wpf) (1.2 cm body length, juveniles) and 12 wpf (2.5 cm body length, adults), showing absence of morphological defects in embryos, onset of spine curvature (tail-up) in juveniles and scoliosis in adults. (B, C) Graph showing the time course of scoliosis appearance (B) in rpgrip1l-/+ incrosses (total 4 clutches, 252 fish) and scoliosis penetrance (C) in adults. (D, E) Micro-computed tomography (?CT) scans of control siblings (D) and rpgrip1l-/- (E) adult fish. Four fish were analyzed for each condition. (F, G) Dorsal and lateral superimposed ?CT views of the spines of one control (yellow) and two rpgrip1l-/- (pink, cyan) adult fish illustrating the 3D spine curvatures in mutants. (H) Schematic representation of the two rescue constructs. (I) Table presenting the number and phenotype of rpgrip1l-/- fish generated for both rescue experiments by stable transgenesis (J) Graph representing scoliosis penetrance in transgenic and non-transgenic rpgrip1l-/- siblings.