Rationale:Anti-N-methyl-D-aspartate (NMDA) receptor encephalitis is an immune-mediated syndrome caused by the production of antibodies against NMDA receptors. As NMDA receptors are important targets of many anesthetic drugs, the perioperative management of patients with anti-NMDA receptor encephalitis is challenging for anesthesiologists.Patient concerns:A 31-year-old woman presented with akinesia and aphasia, which worsened despite steroid therapy.Diagnosis:Anti-NMDA receptor encephalitis associated with ovarian teratoma.Interventions:Laparoscopic ovarian cystectomy was performed under total intravenous anesthesia (TIVA) with peripheral nerve block (PNB).Outcomes:The patient recovered without postoperative complications or any adverse events after surgery.Lessons:Ideal anesthesia for a patient with anti-NMDA receptor encephalitis is still under discussion. We decided to perform TIVA with PNB because the effect of propofol on NMDA receptors is considered less than that of volatile anesthetics; moreover, PNB may reduce the amount of propofol and opioids required for anesthesia. To conclude, TIVA with PNB may be the most appropriate method for anesthesia in a patient with anti-NMDA receptor encephalitis undergoing ovarian cystectomy.