Objective To report a case of Elizabethkingia anopheles (E. anopheles)-associated sepsis and meningitis, review the relevant literatures, and provide reference for the diagnosis and treatment of this infection. Methods Treatment process of a pediatric patient aged 11-year-7-month old who developed post-neurosurgery E. anopheles-associated sepsis and meningitis was analyzed retrospectively. Relevant literature was reviewed to summarize clinical characteristics of patients with E. anopheles-associated sepsis or meningitis. Results The main clinical manifestations of this pediatric patient were fever and abnormal cerebrospinal fluid routine. The cerebrospinal fluid and blood cultures showed E. anopheles. Patient recovered after vancomycin treatment. Including this case, a total of 33 cases were analyzed, with 16 males and 1 unreported gender. The median age of patients was 49 years old (range: 1 day-84 years). Most cases were healthcare-associated infections (62.5%, n=20) with underlying diseases, and 24 cases were cured. Conclusion E. anopheles-associated infections are rare in clinic, but can cause severe infections such as sepsis and meningitis, warranting clinical attention.