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Autoimmune encephalitis (AE) is an inflammatory syndrome of the central nervous system (CNS) triggered by aberrant immune responses against neuronal intracellular (IC-AE) or surface (NS-AE) autoantigens. The resulting neuronal alterations and clinical trajectories differ, with IC-AE often leading to fatal outcomes. Unfortunately, the scarce availability of tissue from AE cases has hampered systematic analyses that would allow an understanding of the pathogenesis underlying neuronal alterations in T cell-mediated AE syndromes. Here, we assembled a cohort comprising both NS-AE (n = 8) and IC-AE (n = 12) from multiple institutions to delineate key histopathological features that distinguish neuronal pathology between IC-AE and NS-AE. In contrast to NS-AE, IC-AE lesions present a prominent neuronal pSTAT1 signature, accompanied by a high proportion of brain-resident memory CD8 + T cells and neurodegenerative GPNMB + phagocytes which show synaptic engulfment with little C3-complement deposition. Our findings highlight distinct histopathological features of IC-AE compared to NS-AE, providing actionable biomarkers for diagnostics and treatment strategies.

Original publication

DOI

10.1007/s00401-025-02882-7

Type

Journal article

Journal

Acta Neuropathol

Publication Date

25/04/2025

Volume

149

Keywords

Neurodegeneration, Neuroinflammation, Phagocytes, Resident memory T cells, Synapses, Humans, Encephalitis, Female, Male, Middle Aged, Neurons, Hashimoto Disease, Adult, STAT1 Transcription Factor, Synapses, Aged, Autoantigens, Brain, CD8-Positive T-Lymphocytes, Young Adult