Three examples of frontal 3D-reconstructions of zebrafish’s spine deformities, classified according to Lenke and compared to corresponding human scoliosis. Left: Reconstructions from RaDiant Dicom Viewer 5.0.0 software (https://www.radiantviewer.com/); Right: Clinical plain radiographs (personal collection of LMH).

Juvenile cfap298^tm304/tm304 zebrafish mutants develop an evolutive thoraco-lumbar curvature of the spine. (A) Frontal views of scannographic reconstructions of 8-week-old non-scoliotic (top) and cfap298^tm304 scoliotic sibling (bottom). (B) Frontal and sagittal view of the same cfap298^tm304 scoliotic fish after scannographic reconstruction showing a 3-dimensional torsion of the spine reminiscent of AIS. (C) Distribution of the frequency of the number of spine curves observed in the sagittal plane of scoliotic (grey) and non-scoliotic (white) animals. The population analyzed was raised from a cross of cfap298^tm304/+ with cfap298^tm304/tm304. Most of cfap298 scoliotic animals display double or triple curves in the frontal plane. (D) Distribution of the Cobb angle of the main curve in cfap298 scoliotic fish depending on the localization of the apex of the main curve. Bars represent the average Cobb angles ± SEM. Each point represents a single fish. Most of the main curve apices are located between T6 and L10 (11 out of 17 fish) (E) Histogram showing the distribution of the number of dislocations observed in the scoliotic population (frequency, %).

cfap298^tm304 mutants exhibit a sexual and a right convexity bias for scoliosis development. (A) Stacked histogram showing the distribution of scoliotic and non-scoliotic phenotypes (frequency, %) in the analyzed cohort according to the gender (males: light grey, females: dark grey). Spinal curves were more prevalent in females (dark grey). (B) Distribution of the Cobb angle of the main curve in cfap298^tm304 scoliotic fish according to the side of the convexity in the frontal plane (right versus left, grey points; each point represents a single fish). The frequency of the convexity of the main curve is represented in blue. Note that most of the curves are biased to the right side (12 out of 17 fishes).