摘要Anti-N-methyl-D-aspartate receptor (NMDAR) encephalitis is caused by reversible neuron dysfunction associated an autoantibody-mediated decrease of NMDAR in the entire brain. A N-methyl-D-aspartate (NMDA) -agonist treatment for anti-NMDAR encephalitis might have a role considering its speciifc mechanism. The authors used D-cycloserine, a partial NMDA-agonist in a refractory case with prolonged intensive care unit duration. A 13-year-old female presented with headache, cognitive deterioration, generalized seizures, coma and hypoventilation with required mechanical ventilation. Anti-NMDAR antibodies were identiifed in cerebrospinal lfuid and serum conifrming anti-NMDAR encephalitis. The patient was refractory to ifrst-line and second-line immunotherapy and removal of ovary teratoma. D-cycloserine was then administered and her symptoms improved gradually and signiifcantly. This is the ifrst reported case in which D-cycloserine was applied to this disease. D-cycloserine might be a potential option as speciifc treatment in anti-NMDAR encephalitis.