Fig. 1 gabra4 mutant behavior and transcript expression in 48 hpf zebrafish: (A) Schematic, modified from Teicher et al. 2025, that illustrates the landmarks used to generate body angles. (B) Schematic of the body angles produced over time during a normal escape response. Beige lines represent the 110º and -110º thresholds for a high amplitude body bend (H.A.B.B.) or C bend. The horizontal line with arrow heads shows the measurement of period length. The vertical line with arrow heads represents body bend amplitude. (C) Radar plot of gabra4 mutant kinematic behavior averages normalized to sibling controls. Average Control values are 100%. M.A., Mean Amplitude; W.I.I., Wavelength Irregularity Index; M.W., Mean Wavelength; Max V., Maximum Velocity; M.V., Mean Velocity; Dur., Duration; H.A.B.B., High Amplitude Body Bends; A.I.I., Amplitude Irregularity. Only Mean Velocity is significantly different compared to controls. **P<0.01 using unpaired Welch’s t-tests. (D-F) Box plots of averages with standard deviation for gabra4 mutants and sibling controls. (D) No significant differences were observed in swimming duration, shown in milliseconds or (E) H.A.B.B. per response. (F) However, gabra4 mutants exhibit an increase in Mean Velocity in millimeters per second. (G-R) Confocal analysis of gabra4 and pkd2l1 expression. (G) Shows expression of gabra4 in the developing brain. The regions outlined by dashed the white boxes are shown at higher magnification in H and I. (J) gabra4 is expressed in a subset of cells in the spinal cord. The region outlined by the dashed box in P is shown at higher magnification in K and L. (M-O) pkd2l1 is expressed in spinal cord CSF-cNs. CSF-cNs consist of two distinct subtypes located around the central canal, a dorsal population (arrowheads) and a ventral population (asterisks). (P-R) Merged channels show extensive colocalization of gabra4 and pkd2l1, indicating that gabra4 is expressed in dorsal and ventral subtypes of CSF-cNs. Scale bars indicate 50μm, 10μm, and 5μm.
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