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Introduction. Minipuberty occurs during the first months of life after the activation of the hypothalamic-pituitary-gonadal axis which causes an increase in gonadotropic and sex hormones. Usually, it does not induce clinically evident physical changes. Studies have shown that minipuberty in extremely premature infants is more pronounced and lasts longer, leading to higher levels of sex hormones induce climically evident in physical changes. Case Report. We present two extremely premature female infants, born at 25 weeks of gestation, with clinically evident physical changes during minipuberty. The first infant presented with vaginal bleeding at the age of 4 months, corrected age of 2 weeks. The vaginal bleeding lasted for two days and stopped spontaneously. The infant also had small glandular breast buds of 1 cm bilaterally, swelling in the pubic region, swollen vulva and clitoris. The second infant presented with swelling in the genital region, suprapubic area and the anterior part of thighs, at the age of 4 months, corrected age of 2 weeks. Both infants had ovarian cysts. In both cases, laboratory tests were consistent with minipuberty. The described changes disappeared gradually and spontaneously. Conclusion. In order to avoid unnecessary testing, clinicians should be aware of possible physical changes during minipuberty in extremely premature infants. Clinical monitoring of these infants is recommended until regression of newly developed physical characteristics.