Effect of rituximab in patients with leucine-rich, glioma-inactivated 1 antibody-associated encephalopathy

JAMA Neurol. 2014 Jul 1;71(7):896-900. doi: 10.1001/jamaneurol.2014.463.

Abstract

Importance: This observational study describes the efficacy and safety of rituximab in 5 patients with voltage-gated potassium channel (VGKC)-complex/leucine-rich, glioma-inactivated 1 (LGI1) antibody-associated encephalopathy. Rituximab is a monoclonal antibody that targets CD20 and is used to treat other neurologic and nonneurologic diseases.

Observations: This case series reports sequential seizure frequencies, modified Rankin Scale scores, and VGKC-complex antibody titers in 5 adult patients (median age, 65 years; range, 48-73 years) treated with rituximab. Median time from symptom onset to rituximab initiation was 414 days (range, 312-851 days). One patient showed a rapid clinical improvement after treatment with rituximab alone and experienced a rituximab-responsive clinical relapse. Another showed possible improvement on neuropsychometric memory indexes after rituximab therapy. In contrast, all patients showed robust responses to treatment with glucocorticoids, intravenous immunoglobulins, and/or plasma exchange at some point in their illness. Treatment with glucocorticoids-less so with intravenous immunoglobulins and plasma exchange-was associated with the most marked reductions in VGKC-complex antibodies. The only patient who did not receive glucocorticoids showed the poorest clinical and serologic responses.

Conclusions and relevance: Rituximab was well tolerated in this predominantly older adult patient population and may be an effective option for some patients with LGI1 antibody-associated encephalopathy. Glucocorticoid therapy appears particularly efficacious. Earlier rituximab administration and randomized trials are required to formally assess efficacy.

Publication types

  • Case Reports
  • Observational Study
  • Research Support, N.I.H., Extramural
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Aged
  • Antibodies, Monoclonal, Murine-Derived / administration & dosage
  • Antibodies, Monoclonal, Murine-Derived / pharmacology*
  • Antigens, CD20 / metabolism
  • Autoantibodies / biosynthesis*
  • Autoantibodies / blood
  • Brain Diseases / drug therapy
  • Brain Diseases / immunology*
  • Female
  • Humans
  • Immunologic Factors / administration & dosage
  • Immunologic Factors / pharmacology*
  • Intracellular Signaling Peptides and Proteins
  • Male
  • Middle Aged
  • Potassium Channels, Voltage-Gated / immunology
  • Proteins / immunology*
  • Retrospective Studies
  • Rituximab
  • Severity of Illness Index
  • Treatment Outcome

Substances

  • Antibodies, Monoclonal, Murine-Derived
  • Antigens, CD20
  • Autoantibodies
  • Immunologic Factors
  • Intracellular Signaling Peptides and Proteins
  • LGI1 protein, human
  • Potassium Channels, Voltage-Gated
  • Proteins
  • Rituximab