Increased production of androgens in the embryonic period leads to virilization of the external genital organs in girls with congenital adrenal hyperplasia (CAH). A significant effect of androgens on the embryonic urogenital sinus can lead to the development of paraurethral glands (PUG) and their differentiation according to the type of tissue of the prostate gland (PG). The presented clinical observations demonstrate MRI diagnostics of PG tissue in girls with CAH and confirm the hypothesis of androgen-dependent PUG. Significant virilization of the external genitalia (Prader IV—V) at birth in both patients indicates high levels of androgens, which created the conditions for the growth of the PUG during fetal development. Decompensation of CAH during puberty, which led to a prolonged period of increasing testosterone levels in the blood of patients, could also lead to further growth of the PUG and visualization of tissue identical PG on MRI. The presence of PG in girls with karyotype 46, XX and CAH is a rare case of thepathological development of the enitourinary tract. Due to the risk of adenocarcinoma and benign prostatic hyperplasia, such patients should undergo annual PSA monitoring and pelvic MRI in case of an increase PSA. Issues related to the causes and frequency of PG visualisation in girls with CAH, as well as recommendations for their treatment, require further study.