Overlapping demyelinating syndromes and anti–N-methyl-D-aspartate receptor encephalitis

Ann Neurol. 2014 Mar;75(3):411-28. doi: 10.1002/ana.24117.

Abstract

Objective: To report the clinical, radiological, and immunological association of demyelinating disorders with anti–Nmethyl- D-aspartate receptor (NMDAR) encephalitis.

Methods: Clinical and radiological analysis was done of a cohort of 691 patients with anti-NMDAR encephalitis. Determination of antibodies to NMDAR, aquaporin-4 (AQP4), and myelin oligodendrocyte glycoprotein (MOG) was performed using brain immunohistochemistry and cell-based assays.

Results: Twenty-three of 691 patients with anti-NMDAR encephalitis had prominent magnetic resonance imaging (MRI) and/or clinical features of demyelination. Group 1 included 12 patients in whom anti-NMDAR encephalitis was preceded or followed by independent episodes of neuromyelitis optica (NMO) spectrum disorder (5 cases, 4 anti-AQP4 positive) or brainstem or multifocal demyelinating syndromes (7 cases, all anti-MOG positive). Group 2 included 11 patients in whom anti-NMDAR encephalitis occurred simultaneously with MRI and symptoms compatible with demyelination (5 AQ4 positive, 2 MOG positive). Group 3 (136 controls) included 50 randomly selected patients with typical anti-NMDAR encephalitis, 56 with NMO, and 30 with multiple sclerosis; NMDAR antibodies were detected only in the 50 anti-NMDAR patients, MOG antibodies in 3 of 50 anti-NMDAR and 1 of 56 NMO patients, and AQP4 antibodies in 48 of 56 NMO and 1 of 50 anti-NMDAR patients (p<0.0001 for all comparisons with Groups 1 and 2). Most patients improved with immunotherapy, but compared with anti-NMDAR encephalitis the demyelinating episodes required more intensive therapy and resulted in more residual deficits. Only 1 of 23 NMDAR patients with signs of demyelination had ovarian teratoma compared with 18 of 50 anti-NMDAR controls (p50.011).

Interpretation: Patients with anti-NMDAR encephalitis may develop concurrent or separate episodes of demyelinating disorders, and conversely patients with NMO or demyelinating disorders with atypical symptoms (eg, dyskinesias, psychosis) may have anti-NMDAR encephalitis.

MeSH terms

  • Adolescent
  • Adult
  • Animals
  • Anti-N-Methyl-D-Aspartate Receptor Encephalitis / complications*
  • Anti-N-Methyl-D-Aspartate Receptor Encephalitis / diagnosis
  • Anti-N-Methyl-D-Aspartate Receptor Encephalitis / immunology
  • Anti-N-Methyl-D-Aspartate Receptor Encephalitis / pathology
  • Aquaporin 4 / immunology
  • Autoantibodies / immunology
  • Brain / immunology
  • Brain / pathology
  • Child
  • Child, Preschool
  • Demyelinating Diseases / complications*
  • Demyelinating Diseases / diagnosis
  • Demyelinating Diseases / immunology
  • Demyelinating Diseases / pathology
  • Female
  • Humans
  • Infant
  • Male
  • Middle Aged
  • Myelin-Oligodendrocyte Glycoprotein / immunology
  • Neuroimaging
  • Rats
  • Receptors, N-Methyl-D-Aspartate / immunology

Substances

  • Aquaporin 4
  • Autoantibodies
  • Myelin-Oligodendrocyte Glycoprotein
  • Receptors, N-Methyl-D-Aspartate