Lrrc71 disruption resulted in male infertility involving mitochondrial sheath and sperm annulus damage. Lrrc71 could directly bind to Septin4 through its N-terminal. Mechanistically, Lrrc71 stabilizes the sperm annulus by preventing the autophagy pathway degradation of Septin4. Collectively, our findings demonstrate that Lrrc71 serves as a central hub regulating sperm motility and annulus integrity, providing insights into the pathogenic mechanisms of infertility caused by human Lrrc71 mutations.