ZFIN ID: ZDB-FIG-200728-41
Troutwine et al., 2020 - The Reissner Fiber Is Highly Dynamic In Vivo and Controls Morphogenesis of the Spine. Current biology : CB   30(12):2353-2362.e3 Full text @ Curr. Biol.
ADDITIONAL FIGURES
PHENOTYPE:
Fish:
Observed In:
Stage Range: Protruding-mouth to Adult

Fig. 1 Hypomorphic Mutations of Scospondin Lead to Progressive Scoliosis in Zebrafish (A–B’) MicroCT images of scospondinstl300/+ (A and A’) and scospondinstl300/stl300 mutant zebrafish (B and B’) at 90 dpf, in both lateral (A and B) and dorsal (A’ and B’) views showing adult-viable scoliosis. (C and D) Bright-field image of the typical straight body of a wild type (C) and atypical axial curvatures observed in homozygous scospondinstl297/stl297 mutant (D) larvae at 13 dpf. Scale bar, 1 mm. (E) Dorsal-ventral axis displacement of the notochord in 5-dpf heterozygous scospondinstl297/+ and scospondinstl297/stl297 mutants (mean + SEM, n = 10 and 23, respectively). (E’) Displacement is the absolute value of the dorsal-ventral (y axis) measured against an origin set at the intersection of the first somite boundary and the dorsal edge of the notochord and propagated along the entire axis at each somite boundary for each animal. (F) Incidence of axial curvatures over developmental time for wild type, MZscospondinstl297, and progeny from heterozygous scospondinstl297/+ × scospondinstl297/stl297 mutant crosses from both female and male homozygotes and progeny from heterozygous scospondinstl297/+ incrosses (mean + SEM; n = 112, 105, 116, 138, and 118 embryos, respectively, pooled from three independent clutches). (G) Schematic representation of zebrafish SCO-spondin protein, demarking the location of the scospondin alleles causing scoliosis in zebrafish reported in this study. Boxes represent conserved motifs; legend in Figure S1. (H and J) Protein alignments (Clustal-W) of SCO-spondin protein showing the sequence surrounding the amino acid residues (yellow highlight) affected by the scospondinstl297 (H) and the scospondinstl300 (J) mutations. (I) A homology model of Danio rerio SCO-spondin threaded onto the LDLrA domain derived from an LDL receptor structure (PDB: 6byv). The predicted disulfide bond and labeled cysteine (Cys) residues are highlighted in (H) and (I). See also Figures S1 and S2.

Gene Expression Details No data available
Antibody Labeling Details No data available
Phenotype Details
Fish Conditions Stage Phenotype
sspostl297/+ standard conditions Protruding-mouth - Days 14-20 whole organism axis curved, abnormal
sspostl297/stl297 standard conditions Protruding-mouth whole organism axis curved, abnormal
sspostl297/stl297 standard conditions Protruding-mouth - Days 14-20 whole organism axis curved, abnormal
sspostl297/stl297 standard conditions Protruding-mouth - Days 14-20 whole organism axis curved, abnormal
sspostl297/stl297 standard conditions Protruding-mouth - Days 14-20 whole organism axis curved, abnormal
Day 5 whole organism dorsal-ventral axis curved, abnormal
Days 7-13 whole organism dorsal-ventral axis curved, abnormal
Days 7-13 whole organism medial-lateral axis curved, abnormal
Adult vertebra morphology, normal
Adult vertebral column bone development process quality, normal
Adult vertebral column cartilage development process quality, normal
Adult whole organism dorsal-ventral axis curved, abnormal
Adult whole organism medial-lateral axis curved, abnormal
sspostl300/stl300 standard conditions Protruding-mouth whole organism axis curved, abnormal
Adult vertebra morphology, normal
Adult vertebral column bone development process quality, normal
Adult vertebral column cartilage development process quality, normal
Adult whole organism dorsal-ventral axis curved, abnormal
Adult whole organism medial-lateral axis curved, abnormal
Acknowledgments:
This image is the copyrighted work of the attributed author or publisher, and ZFIN has permission only to display this image to its users. Additional permissions should be obtained from the applicable author or publisher of the image. Full text @ Curr. Biol.