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Delayed NMDA Receptor Encephalitis Relapse in a Male Patient.

Here, we report a case of a 36 year old man with antibody-confirmed NMDA receptor 17 encephalitis who was admitted at age 15 with a similar presentation, representing a delay to first 18 relapse of 21 years. Our patient presented at age 36 with a 10-day history of progressive word 19 finding difficulties and paranoid ideation, preceded by URTI symptoms. On admission 20 examination, he was bradyphrenic with expressive aphasia, short term memory impairment, and 21 executive dysfunction. He had left beating gaze-evoked nystagmus and orofacial dyskinesias, 22 but no other cranial nerve, sensorimotor, or cerebellar abnormalities. Empiric ceftriaxone and 23 acyclovir were started. A lumbar puncture showed elevated protein (1395 mg/L) and WBC (56 24 X10^6 cells/L82% neutrophils). Viral PCR was negative. Contrast MRI head was 25 unremarkable. EEG showed changes suggestive of extreme delta brush pattern (Figure 1). He 26 progressed to frank psychosis and florid agitation by post-admission day 2, and autonomic 27 disturbance with altered temperature regulation and sinus tachycardia by day 7. IVIG (2g/kg) 28 was started at day 5, followed by a 5 day course of IV methylprednisolone at day 10 given no 29 early response to IVIG. IVIG was initiated before steroids due to concern about exacerbating 30 psychosis. NMDA receptor antibodies returned strongly positive in the serum and CSF. 31 Neoplastic screen (CT of the chest, abdomen, and pelvis, with testicular ultrasound) was 32 negative. He continued to deteriorate to a nonverbal state by day 14. Due to lack of response to 33 immunotherapy, plasma exchange (PLEX) was performed at day 20, and Rituximab (1g IV X 2 34 doses 2 weeks apart) started after PLEX at post-admission day 30. His neurological status 35 improved markedly over 3 weeks following 5 cycles of PLEX. On discharge at day 50, he had 36 marked improvement with a Montreal Cognitive Assessment (MoCA) score of 25/30. After 37