Fig. 2

cfap298 is required for CSF transport down the rostrocaudal axis and activity of sensory CSF-contacting neurons. Representative images of 28 hpf a cfap298^+/+ and b cfap298^tm304/tm304 embryos 60 min after injection of fluorescent beads into the hindbrain ventricle (labeled by the black line), the bead front is indicated by an arrowhead in a. Scale: 250 µm. c Quantification of dye front experiments. (n = 18 straight cfap298^+/+ and cfap298^tm304/+ embryos, n = 17 curled cfap298^tm304/tm304 embryos). Each point represents one embryo. Error bars represent s.e.m, *p < 0.05, **p < 0.01. d Sample traces of calcium activity in CSF-cNs labeled in Tg(pkd2l1:GCaMP5G) at 25 hpf in cfap298^+/+, cfap298^tm304/+, and cfap298^tm304/tm304. Traces represent cells with the median integral values taken from all embryos. Scale: 30 s, 30% ΔF/F. e Normalized and integrated calcium activity per embryo. Each point represents the average activity across all CSF-contacting neurons for one embryo. cfap298^m304/tm304 embryos (n = 11 embryos, 165 cells) had significantly reduced activity compared to wild type (n = 8 embryos, 73 cells; one-way ANOVA, t = 3.7, p = 1.7 × 10^‒3, α = 0.0167 following Bonferroni correction for multiple testing). The central mark on the box plot indicates the median, the bottom and top edges of the box indicate the 25th and 75th percentiles. The whiskers extend to the most extreme data points not considered outliers, and the outlier is identified with a “+” symbol