Electroencephalogram (EEG) abnormalities are very common in anti-N-methyl-d-aspartate receptor (anti-NMDAR) encephalitis. Extreme delta brush (EDB) is a distinctive EEG pattern that is can be suggestive of the diagnosis; however, the etiology of the EDB remains unclear. Furthermore, there is question with regard to its ictal or interictal nature. We report a 20-year-old woman with anti-NMDAR encephalitis whose serial video-EEG monitoring was obtained at 2, 2.5, 4, and 6 months after admission. There was a long-standing EDB lasting up to several hours, with no evolution in frequency, amplitude, or morphology, and without clear association her frequent orofacial dyskinesia. Intravenous benzodiazepine administrations did not change the EDB pattern. As her clinical symptoms improved, the EDB gradually became less prominent and less frequent, with complete resolution at 6 months after admission. These findings suggest that EDB is more likely a marker of the severity of the disease in contrast to an epileptic seizure and is useful for diagnosis and monitoring of treatment response in conjunction with clinical improvement.