Aberrations of CATSPER genes, some of which were shown to result in a loss of CatSper function, are associated with male infertility (Avidan et al., 2003; Avenarius et al., 2009; Hildebrand et al., 2010; Smith et al., 2013; Jaiswal et al., 2014; Williams et al., 2015; Brown et al., 2018; Luo et al., 2019; Schiffer et al., 2020; Wang et al., 2020), indicating that CatSper serves as a central signaling knot in human sperm. Here, CATSPER1 is linked to male infertility.