Objectives: This is a retrospective observational study conducted in one of the largest clinical center of neurosurgery in China. Our aim was to determine the epidemiological characteristics of carbapenem-resistant Enterobacteriaceae (CRE) related meningitis/encephalitis and to elucidate the risk factors for CRE neurosurgical infections. Patients and methods: We performed a retrospective study between January 2012 and December 2017 of patients who underwent neurosurgery. The medical records of each patient were reviewed, and 20 clinical variables on risk factors were extracted and evaluated by Multivariate logistic analysis for CRE-meningitis/encephalitis. Results: In 2012–2017, the positive rate of neurosurgical meningitis/encephalitis was 7.9% (2947/29605), Enterobacteriaceae accounted for 6.3% (185/2947) of all bacterial infections. Totally, 133 Enterobacteriaceae include 26 CRE isolates were available in this study. Of them, Univariate analysis showed that the risk factors of CRE meningitis were ventilator, bacteremia, Intensive Care Unit (ICU) admission, hospital acquired pneumonia and mortality attribute to infection. Multivariate logistic analysis showed that hospital acquired pneumonia and mortality attribute to infection were independent risk factors for CRE meningitis. Conclusion: CRE is one of the most serious drug-resistant bacteria published by World Health Organization (WHO) in 2016, and meningitis/encephalitis caused by CRE is an important sign of the failure of the neurosurgery, which demands the physician's immediate attention. © 2019 Japanese Society of Chemotherapy and The Japanese Association for Infectious Diseases