TCTE1 is a conserved component of the dynein regulatory complex and is required for motility and metabolism in mouse spermatozoa

Significance Infertility is a global problem that afflicts 15% of couples, and in 50% of cases, the attributing factor is linked to men. Among these infertile men, 18% specifically exhibit decreased motility of sperm (asthenozoospermia). Sperm motility is dependent on the formation and functioning of the flagellum, a modified cilium used for locomotion. Cilia are present in almost every cell of vertebrates and are essential for proper organ functioning. Defects in cilia formation lead to severe syndromic diseases, termed ciliopathies, affecting numerous tissues (e.g., polycystic kidney disease), wherein male infertility is often comorbid. Advances in mouse genetics implicate several genes responsible for ciliopathies observed in humans. Here, we identify a nonsyndromic flagellum protein, TCTE1, that is required for sperm motility in mice.