Abstract: Objective To discuss the early recognition and treatment of group B streptococcus (GBS) infection caused neonatal meningitis and subdural effusion. Method The onset, clinical manifestations, diagnosis and treatment process were retrospectively analyzed in one case of typical GBS infection caused neonatal meningitis and subdural effusion. Results The subject was late-onset GBS infection, with insidious onset, rapid progress, slow clinical recovery, and highly sensitive to vancomycin. During the treatment, the subject had relapses. The subdural effusion had been found. After extension of vancomycin treatment, the subject recovered. Conclusions The late onset GBS infection should be taken seriously in clinical, pay attention to the complications such as purulent meningitis, subdural effusion, hydrocephalus, and be early treated with adequate and effective antibiotics.