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Uterine leiomyoma represent a common finding in women at fertile age, being usually responsible for the apparition of metrorhagia and pelvic pain; in certain cases presenting large uterine fibroids, tumoral masses can be encountered at the clinical abdominal exam. However, in certain cases they can be responsible for the apparition of urinary signs such dysuria and pollakiuria due to their compression on the urinary bladder. The aim of this paper is to report the case of a 56-year-old patient who presented for oligo-anuria and bilateral ureteral hydronephrosis and who was diagnosed with a large intra-istmic leiomyoma with vaginal development which induced the development of acute urinary retention. The patient was submitted to surgery, a total hysterectomy with bilateral adnexectomy being performed.