Objective The aim of this study was to identify risk factors for early-onset GBS (EOGBS) disease in neonates of mothers with negative antenatal screening. Study Design We performed a retrospective cohort study of neonates born to mothers with negative antenatal GBS screening between 2002–2012. Our primary outcome was EOGBS infection. We used multivariable logistic regression to assess factors associated with EOGBS. Results EOGBS was confirmed in 492 of the 179,818 neonates that met the study inclusion criteria. Risk factors for EOGBS included black race (reference: white, odds ratio [OR] =1.81 [95% confidence interval; 1.43, 2.31]), maternal age <18 years (reference: >35 years, OR=2.63 [1.54, 4.51]), and maternal age 18–35 years (reference: >35 years, OR=1.94 [1.30, 2.88]). Conclusion Maternal age <18 years and black race were the strongest predictors of EOGBS. Further research investigating contributors to the discordance between screening results and neonatal outcomes in these populations is needed.