Intraventricular empyema is a complication of meningitis and associated with concomitant hydrocephalus due to blockage of CSF pathway. If not recognized and treated early, it can lead to significant morbidity and mortality. Streptococcus pneumoniae, Neisseria meningitidis, and Staphylococcus aureus are most commonly reported in infants along with other rare pathogens. Burkholderia cepacia complex previously called Pseudomonas is a rare phytopathogenic, saprophytic Gram-negative bacilli causing opportunistic infections in hospitalized and immunocompromised patients. It has been reported to cause septicemia in few case series. However, CNS infections are rarely reported in few case reports. We present here a preterm neonate born unexpectedly outside the hospital presented with the complication of intraventricular empyema due to Burkholderia infection and the subsequent course of management.