In this article, a 6-month-old infant with cystic dysplasia of the testis (CDT) is presented. Although different genitourinary abnormalities associated with CDT have been reported, this is the first case of the association of CDT with penoscrotal hypospadias, unilateral undescended testicle, bilateral scrotal hernia, small and hypocompliant bladder and urethral obstruction. Orchiectomy was carried out for the treatment of this rare cause of testicular cystic mass.