During a 6-year period, the prospective interpretations of routine screening cranial ultrasound on 1453 premature infants indicated frontal periventricular cysts in 7 infants without other intracranial abnormalities during the first 24 hours after birth. The medical records of these 7 infants and their mothers and a developmental assessment at age 1 year were also reviewed. The cysts were predominantly single and unilateral and ranged in size from 2 to 11 mm. One infant had bilateral subependymal cysts as well as frontal periventricular cysts. All 7 infants before 1 year of age demonstrated mild neurological deficiencies. However, 6 of 7 demonstrated partial or complete resolution of their neurological impairment by the end of 1 year. One infant was lost to clinical follow-up. The presence of these frontal periventricular cysts in the first 24 hours after birth suggests an in-utero event, such as antepartum hemorrhagic infarction, as their etiology.