<i>In vivo</i> CAPZA1 deletion was induced via adeno-associated virus (AAV)-mediated CRISPR-Cas9 delivery into mouse testes, and subsequent sperm motility, protein expression, and ultrastructure were evaluated.<h4>Results</h4>A rare homozygous missense mutation in CAPZA1 (c.11T>C, p.Phe4Ser) was first identified by WES in the proband of an infertile family and was subsequently detected by Sanger sequencing in 3 of 20 asthenozoospermic patients. This evidence concerns the gene CAPZA1 and Infertility.