In this study, we identified novel CFAP57 gene mutations in two unrelated infertile males: the first patient (P1) carrying a homozygous nonsense mutation [NM_001378189.1: exon 20: c.3250 C >T (p.R1084X), NM_001195831.3:exon 21:c. Here, CFAP57 is linked to Infertility.