Background: Paraprostatic cysts are large structures that develop between the prostate gland and urinary bladder, usually in older, intact dogs. Their incidence is reported to be 1.1-5.3% in dogs with prostatic disease. The aetiology of paraprostatic cysts is not fully understood, but they are believed to develop from the uterus masculinus. Whereas the uterus masculinus has been reported to communicate with the urethra in men and horses, no communication between the uterus masculinus and urethra has been identified in dogs. Case presentation: An entire male dog was presented with a bloody discharge from its penis and tenesmus of 5 days' duration. A diagnosis of cystic uterus masculinus was made on the basis of the findings of abdominal ultrasonography and histopathology of tissues obtained during an exploratory laparotomy. In addition, a Sertoli cell tumour affecting both testes was diagnosed following scrotal castration. The cystic uterus masculinus was completely resected, after which the tenesmus and bloody discharge resolved. Thus, cystic uterus masculinus should be considered as a differential diagnosis for a paraprostatic cyst when such a lesion develops as part of the feminising effect of a Sertoli cell tumour. Conclusions: Cystic uterus masculinus should be considered as a differential diagnosis for tenesmus and penile discharge, and for structures resembling paraprostatic cysts. This case report confirms that a uterus masculinus can communicate with the urethra in dogs, as in other species, and demonstrates endocrine responsiveness, manifesting as epithelial and glandular metaplasia and mucus production, with the potential for subsequent infection.