Serratia marcescens is a recognized nosocomial opportunistic pathogen but rarely caused central nervous system infection especially in the neonates. Outbreaks have been documented in the neonatal intensive care units (NICU) and a higher incidence among those with surgical procedures. This review aims to describe a neonate with nonleaking lumbosacral myelomeningocele presenting with multiple pyogenic brain abscesses caused by S. marcescens admitted in a NICU. This review also presents a concise literature review discussing the potential risk factor involved, diagnostic measures and therapeutic possibilities. We present a neonate with Chiari II malformation admitted in the NICU developing S. marcescens ventriculitis after a lumbosacral myelomeningocele repair. With an empiric treatment of meropenem for one week, repeat ventricular cerebrospinal fluid analysis worsened and developed cerebral abscess as detected using cranial ultrasound. Ciprofloxacin was added and completed for six weeks with improved neurologic status. On a 6-month follow-up, sensorineural hearing loss, focal epilepsy and developmental delay were documented. A systematic review showed that prematurity and NICU outbreaks were among the most common risk factors for the central nervous system involvement of S. marcescens. Meropenem remains to be the antibiotic of choice adjunct with timely neurosurgical intervention. Brain abscess showed the worst prognosis among the neurologic sequelae.