Zinner综合征是一种先天性泌尿生殖系胚胎畸形，临床罕见，发病隐匿，不易诊断，多于性活跃期被发现，首选确诊方法是MRI。对于无症状者多采取保守治疗，有症状患者可通过腹腔镜或机器人手术治疗。对于合并不育的患者如考虑有射精管梗阻，可行经尿道射精管切开术，有助于提高精子活力及数量，增加精液量，但部分患者术后仍为无精症，病因有待进一步研究。本文报道1例Zinner综合征合并输尿管异位开口于精囊囊肿的不育症患者，经手术治疗精液质量得到明显改善。“,”Zinner syndrome is a congenital genitourinary embryo malformation, which is clinically rare, difficult to diagnose, and usually found in the sexually active stage. MRI is the preferred method for diagnosis. Conservative treatment was usually conducted for most asymptomatic patients. Symptomatic patients can be treated by laparoscopic or robotic surgery. For patients with infertility complicated with ejaculatory duct obstruction, transurethral resection of the ejaculatory duct is feasible, which contributes to improving sperm vitality and quantity and increasing semen volume. However, some patients still have azoospermia after surgery. The etiology is needed further studied. A case of infertility with Zinner syndrome and ectopic ureteral opening in seminal vesicle cyst is reported.