Figure 5.
FAP256A/B-KO ciliary tips show multiple structural defects. (A) Slices of WT and FAP256A/B-KO tomograms. The arrowhead points to the end of the CP microtubules to highlight the lack of cap in mutant tomograms. (B) Tomogram slice of FAP256A/B-KO cilium along with cross-section (dotted line). The arrowheads indicate the densities inside the CP microtubules that correspond to the plugs. (C) Tomogram slices from the same FAP256A/B-KO cilia in B. The arrowheads indicate the end of the CP microtubules. (D) Quantification of tip CP length in WT (n = 38) and FAP256A/B-KO (n = 50) cilia. Each dot represents one CP. ****P value < 0.0001, Mann‒Whitney test. Black line indicates the median. (E) Graph of the distance between C1 and C2 microtubules. The reference on the x-axis is the middle of the narrowing zone. The average is shown with a solid black line. Standard deviation is shown in brown (n = 6 cilia). For comparison, the corresponding CU428 data are indicated by a dotted line. (F) Length of A-tubules numbered from shortest to longest. The brown dotted line shows a linear trend line going through all points. Error bars show the standard deviation. n = 8 cilia. For comparison, the corresponding CU428 data are indicated by a dotted line. (G) Tomogram slice of a FAP256A/B-KO mutant cilium. The arrowhead points to a bent singlet A-tubule. (H) Graph showing the distance between pairs of A-tubule singlets in the ciliary tip (n = 6 cilia). Standard deviation is shown in brown. For comparison, the corresponding CU428 data are indicated by a dotted line.

FAP256A/B-KO ciliary tips show multiple structural defects. (A) Slices of WT and FAP256A/B-KO tomograms. The arrowhead points to the end of the CP microtubules to highlight the lack of cap in mutant tomograms. (B) Tomogram slice of FAP256A/B-KO cilium along with cross-section (dotted line). The arrowheads indicate the densities inside the CP microtubules that correspond to the plugs. (C) Tomogram slices from the same FAP256A/B-KO cilia in B. The arrowheads indicate the end of the CP microtubules. (D) Quantification of tip CP length in WT (n = 38) and FAP256A/B-KO (n = 50) cilia. Each dot represents one CP. ****P value < 0.0001, Mann‒Whitney test. Black line indicates the median. (E) Graph of the distance between C1 and C2 microtubules. The reference on the x-axis is the middle of the narrowing zone. The average is shown with a solid black line. Standard deviation is shown in brown (n = 6 cilia). For comparison, the corresponding CU428 data are indicated by a dotted line. (F) Length of A-tubules numbered from shortest to longest. The brown dotted line shows a linear trend line going through all points. Error bars show the standard deviation. n = 8 cilia. For comparison, the corresponding CU428 data are indicated by a dotted line. (G) Tomogram slice of a FAP256A/B-KO mutant cilium. The arrowhead points to a bent singlet A-tubule. (H) Graph showing the distance between pairs of A-tubule singlets in the ciliary tip (n = 6 cilia). Standard deviation is shown in brown. For comparison, the corresponding CU428 data are indicated by a dotted line.

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