Background: Previous studies of neonatal cerebral venous sinus thrombosis (CVST) have focused on term infants, and studies of preterm infants are lacking. In this study, we examined the clinical and radiological features, treatment and outcome of CVST in preterm infants. Methods: This was a retrospective cohort study of preterm infants (gestational age <37 weeks) with radiologically confirmed CVST. All MRI/MRV and CT/CTV scans were re-reviewed. Clinical and radiological data were analysed using descriptive statistics, ANOVA and chi-square tests. Results: A total of 26 preterm infants with CVST were included. Of these, 65% were late preterm, 27% very preterm and 8% extreme preterm. Most (73%) were symptomatic at presentation with seizures or abnormal exam. Transverse (85%) and superior sagittal (42%) sinus were common sites of thrombosis. Parenchymal brain injury was predominantly periventricular (35%) and deep white matter (31%) in location. Intraventricular hemorrhage occurred in 46%. Most infants (69%) were treated with anticoagulation. None of the treated infants had hemorrhagic complications. Outcome at follow-up ranged from no impairment (50%), mild impairment (25%) and severe impairment (25%). Conclusions: Preterm infants with CVST are often symptomatic and have white matter brain lesions. Anticoagulation treatment of preterm CVST appeared to be safe and was not associated with hemorrhagic complications.