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