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Clinico-pathological correlation in unilateral cortical encephalitis treated with tocilizumab after first-line

Samir Alkabie1, Samer Ali2, Saeed Asiry2

  • 1Department of Neurology, Donald and Barbara Zucker School of Medicine at Hofstra/Northwell, Lenox Hill Hospital, New York, NY, United States.

Journal of Neuroimmunology
|September 28, 2025
PubMed

Insights

Tocilizumab effectively treated a rare case of unilateral cortical encephalitis after standard therapies failed. This interleukin-6 receptor blocker highlights immune mechanisms in autoimmune encephalitis.

Area of Science:

  • Neuroimmunology
  • Neurology
  • Immunology

Background:

  • Unilateral cortical encephalitis is a rare neurological condition.
  • Standard treatments may fail, necessitating alternative therapeutic strategies.
  • Understanding immune mechanisms is crucial for guiding treatment in autoimmune encephalitis.

Purpose of the Study:

  • To characterize the clinico-pathologic findings of a rare unilateral cortical encephalitis case.
  • To evaluate the therapeutic response to tocilizumab after first-line treatment failure.
  • To explore potential immune mechanisms involved in the disease pathogenesis.

Main Methods:

  • Case report detailing a 49-year-old male patient.
  • Clinical presentation: fever, seizure, hemicortical deficits.
  • Diagnostic workup included MRI, 18F-FDG-PET, extensive metabolic/infectious/malignancy screening, and neural autoantibody testing.
  • Brain biopsy revealed microglial activation, BBB disruption, and T-cell infiltrates.
  • Treatment response assessed after tocilizumab administration following failed pulse steroids and IVIG.

Main Results:

  • Patient presented with acute unihemispheric encephalitic syndrome with MRI and PET evidence of cortical inflammation.
  • Extensive workup was negative for common causes; neural autoantibodies were absent.
  • Initial treatments (steroids, IVIG) were ineffective.
  • Tocilizumab combined with pulse steroids led to significant recovery in cognition, speech, gait, and resolution of imaging abnormalities.
  • Pathology showed microglial activation, BBB disruption, and T-cell infiltrates, with elevated IL-18.

Conclusions:

  • Tocilizumab, an IL-6 receptor blocker, demonstrated efficacy in this rare encephalitis case.
  • The rationale for tocilizumab involved IL-6's role in T-cell differentiation and CNS inflammation.
  • This case underscores the importance of clinico-pathologic correlation and cytokine profiling for selecting immunotherapy in antibody-negative autoimmune encephalitis.
Abstract

Keywords:
Antibody- negativeAutoimmune encephalitisNeuropathologyTocilizumabUnilateral cortical inflammation

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