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. 2025 Jul 16;12(8):005679.
doi: 10.12890/2025_005679. eCollection 2025.

Anti-N-Methyl-D-Aspartate Receptor Encephalitis Presenting as Isolated First-Episode Psychosis in A 16-Year-Old Girl, Complicated by Neuroleptic Malignant Syndrome

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Anti-N-Methyl-D-Aspartate Receptor Encephalitis Presenting as Isolated First-Episode Psychosis in A 16-Year-Old Girl, Complicated by Neuroleptic Malignant Syndrome

Ghizlane Khaloui et al. Eur J Case Rep Intern Med. .

Abstract

Anti-N-methyl-D-aspartate receptor (anti-NMDAR) encephalitis is a rare but increasingly recognized autoimmune disorder that predominantly affects adolescents and young adults. It often presents with prominent psychiatric symptoms at onset, leading to frequent misdiagnosis as a primary psychiatric disorder. We report the case of a 16-year-old girl who presented with acute psychiatric symptoms, including mood lability, hallucinations, and aggression, initially diagnosed as first-episode psychosis. Her condition rapidly deteriorated under antipsychotic treatment, progressing to neuroleptic malignant syndrome, followed by the emergence of cerebellar ataxia, which raised strong suspicion of an underlying organic aetiology. Subsequent investigations confirmed the diagnosis of anti-NMDAR encephalitis. The patient was successfully treated with intravenous immunoglobulin, achieving complete recovery. This case underscores the diagnostic challenges posed by isolated psychiatric presentations of anti-NMDAR encephalitis in adolescents. Early recognition and prompt initiation of immunotherapy are crucial for favourable outcomes, particularly in cases presenting with antipsychotic intolerance or atypical clinical progression.

Learning points: Anti-N-methyl-D-aspartate receptor encephalitis can initially present as an isolated psychiatric syndrome mimicking primary psychosis, particularly in adolescents, often leading to misdiagnosis.The development of neuroleptic malignant syndrome during a first-episode psychosis, occurring rapidly and at low antipsychotic doses, should raise suspicion of an organic aetiology, notably autoimmune encephalitis.Magnetic resonance imaging, electroencephalogram, and cerebrospinal fluid analysis are essential to distinguish autoimmune encephalitis from primary psychiatric disorders.

Keywords: Anti-N-methyl-D-aspartate receptor encephalitis; adolescent; first-episode psychosis; neuroleptic malignant syndrome.

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Conflict of interest statement

Conflicts of Interests: The Authors declare that there are no competing interests.

Figures

Figure 1
Figure 1
Brain magnetic resonance imaging T2-weighted sequence revealed A) bilateral cerebellar hyperintensity, more pronounced on the left side (arrows). The diffusion-weighted imaging sequence showed B) diffusion hyperintensity in these abnormalities, with corresponding low signal on the apparent diffusion coefficient map (C), consistent with diffusion restriction (arrowheads).

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