In summary, we identified a compound heterozygous mutation in TDRD9 (c.958delC, p. His 320Ilefs*28; c.1115 + 3A > G) in a Chinese male patient with oligozoospermia and confirmed that these two mutations are possible causative factors for oligozoospermia, leading to male infertility. Here, TDRD9 is linked to male infertility.