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Autosomal dominant polycystic kidney disease (ADPKD) is the most common inherited renal disease, half of which has family history, although the exact etiology remains unknown.To date, ADPKD, a multisystem disorder without effective preventive and therapeutic means, has been shown to be harmful to human health.Recently, severe oligoasthenozoospermia, azoospermia, necrospermia,immotile sperm, seminal vesicle cysts, and ejaculatory duct cysts have been found in some patients with ADPKD, with its specific mechanisms enigmatic, which may lead to male infertility.Structural abnormalities of spermatozoa, defects of polycystin as well as mutations of PKD1 and/or PKD2 are probably the main causes for the higher incidence of abnormal semen parameters in ADPKD.We herein present a patient with ADPKD complicated with seminal vesicle and epidymal cysts, which leads to severe oligoasthenozoospermia and hematospermia.We review in detail the current literatures, in order to explore the correlationship between ADPKD and male infertility, meanwhile, inform clinicians caring for ADPKD patients accompany with male reproductive aspects that confront affected individuals.