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目的：提高对附睾乳头状囊腺瘤的认识及诊治水平。方法回顾性分析1例双侧附睾乳头状囊腺瘤的临床特点、影像及病理表现，并进行文献复习。结果一例31岁的男性患者，间断性双侧阴囊坠痛不适1年，入院查体：两侧附睾增大于双侧附睾头分别可触及约5mm大小肿块，B超未给出明确诊断，术后病检确诊双侧附睾乳头状囊腺瘤，随访至今未见明确林岛综合征（Yon Hippel-Lindau Syndrome, VHL综合征）。结论本病确诊来自于病理学检查，附睾乳头状囊腺瘤为良性，手术是治疗附睾肿瘤的首选方法。“,”Objective To emphasize the importance of the early diagnosis and treatment of epididymal papillary of cystadenoma. Methods A case of Bilateral epididymal papillary of cystadenoma including the clinical manifestations image and pathology were reviewed. Results A 31-year-old male presented with discontinuous pain scrotum for one year. two epididymises become bigger, which be found a 5mm lump in them respectively. Ultrasonography of the scrotum did not reveal the truth and pathology after operation confirm abilateral papillary cystadenoma. There was no evidence of von Hippel-Lindau syndrome after diagnosis during the follow-up. Conclusion The diagnosis of epididymal papillary of cystadenoma was confirmed by pathological analysis, and it is benign mass. Surgery is the prime treatment for it.