Background: Neonatal cerebral sinovenous thrombosis (CSVT) can lead to severe brain injury and long-term neurodevelopmental impairments. Previous studies of neonatal CSVT have mainly included term infants. In this study, we examined the clinical and radiological features, treatment and outcome of CSVT in preterm infants. Methods: This was a retrospective cohort study of preterm infants born <37 weeks with radiologically confirmed CSVT. 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 CSVT were included. Of these, 65% were late preterm, 27% very preterm and 8% extreme preterm. Most were symptomatic (seizures 50%, abnormal exam 50%). Radiological features included transverse sinus (85%) and sagittal sinus thrombosis (42%), intraventricular hemorrhage (42%) and venous infarction (19%). Most preterm infants with CSVT (69%) were treated with anticoagulation. Anticoagulation was not associated with new or worsening intracranial hemorrhage. Outcome at follow-up ranged from no impairment (39%), mild impairment (19%), severe impairment (19%) and death (23%). Conclusions: Preterm infants with CSVT are often symptomatic and present with a distinct pattern of brain injury. Anticoagulation treatment of preterm CSVT appeared to be safe. Further studies and treatment guidelines for preterm CSVT are needed.