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. 2019 Jun 20;2:226. doi: 10.1038/s42003-019-0462-y

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© The Author(s) 2019

Open Access This article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The images or other third party material in this article are included in the article’s Creative Commons license, unless indicated otherwise in a credit line to the material. If material is not included in the article’s Creative Commons license and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this license, visit http://creativecommons.org/licenses/by/4.0/.

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Fig. 1 — Generation and phenotypes of Efcab1 mutant mice. a Function of calaxin in the propagation of flagellar and ciliary waveforms. Typical waveforms of Ciona sperm flagella and sea urchin embryonic cilia in normal and calaxin-inhibited conditions are shown. Modified from previous publications^30,32. b Immunoblot analysis of Efcab1^−/− sperm by anti-Efcab1 and anti-acetylated-α-tubulin antibodies. c Immunofluorescence analysis of Efcab1^−/− sperm flagella, trachea and ependymal cilia by anti-Efcab1 and anti-acetylated-α-tubulin antibodies. Scale bar, 50 μm (top) and 25 μm (bottom left and right). d Efcab1^−/− mouse showing hydrocephalus (upper; head morphology, scale bar = 1 cm, lower; 5 µm paraffin section stained with hematoxylin-eosin) and e situs inversus. LV, lateral ventricle; Lv, liver; Sp, spleen. f Percentage of Efcab1^−/− mice with hydrocephalus and/or situs inversus. N = 130 (71; male, 59; female). g Average litter size from parents of different genotypes. h The rate of in vitro fertilization of eggs with epididymal sperm collected from Efcab1^+/+ and Efcab1^−/− mice. Numbers in parentheses indicate the number of eggs examined from triplicate experiments. i Number of embryos at E8, E14 and E16 after mating with Efcab1^+/+ or Efcab1^−/− male mice. j The ratio between heart and body weight in Efcab1^+/+ and Efcab1^−/− mice. k Comparison of lateral ventricle area between Efcab1^+/+ and Efcab1^−/− mice. l Survival rate at 10 weeks. N = 46 (Efcab1^+/+ and Efcab1^+/−) and N = 198 (Efcab1^−/−). Values indicate mean ± SEM. *p < 0.05, ***p < 0.001 vs. Efcab1^+/+ (Student’s t-test)