Objective: To examine the effect of Coagulase-negative Staphylococcus (CoNS) sepsis in preterm infants on the neonatal and neurodevelopmental outcomes at 36 months corrected age (CA). Design: A retrospective cohort study. Subjects: All preterm infants with gestational age ≤ 28 weeks. Results: A total of 105 eligible infants were exposed to CoNS sepsis between 1995 and 2008. Infants exposed to CoNS sepsis were less mature (25.9 ± 1.7 vs. 26.2 ± 1.4, p=0.04), had increased risk of retinopathy of prematurity (ROP) (adjusted RR 1.32; 95% CI 1.11 – 1.54), and were more likely to stay longer in the neonatal intensive care unit. Multivariable logistic regression analysis revealed CoNS sepsis is an independent predictor for cognitive delay (adjusted RR 1.98; 95% CI 1.01 – 3.63). Conclusions: CoNS sepsis in preterm infants is associated with increased risk for ROP in the neonatal period and for cognitive delay at 36 months CA.