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Cognitive & Behavioral Neurology:
March 2009 - Volume 22 - Issue 1 - pp 63-66
doi: 10.1097/WNN.0b013e318190d195
Case Reports

Successful Treatment of Acute Autoimmune Limbic Encephalitis With Negative VGKC and NMDAR Antibodies

Modoni, Anna MD, PhD*; Masciullo, Marcella MD*; Spinelli, Pietro MD*; Marra, Camillo MD, PhD*; Tartaglione, Tommaso MD†; Andreetta, Francesca PhD‡; Tonali, Pietro MD* §; Silvestri, Gabriella MD, PhD* §

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Abstract

Objective: To describe a case of acute nonherpetic limbic encephalitis (LE) with negative testing for antibodies directed against onconeuronal and cell membrane antigens, including voltage-gated potassium channels and N-methyl-D-aspartate receptor, that showed a dramatic response to immune therapy.

Materials and Methods: A 30-year-old woman manifested generalized seizures, altered consciousness, and memory impairment shortly after a prodromal viral illness. Few days later the patient developed a drug-resistant epileptic status.

Results: Electroencephalograph showed bitemporal slowing and paroxysmal slow wave bursts. Brain magnetic resonance imaging showed bilateral swelling in the medial temporal lobes. Cerebrospinal fluid analysis ruled out viral etiologies. A diagnostic search for cancer, including serum testing for known onconeuronal antibodies proved negative. Screening for cell membrane antigen antibodies, including voltage-gated potassium channels and N-methyl-D-aspartate receptor, was also negative. Suspecting an autoimmune etiology, we started an immunomodulatory treatment with intravenous immunoglobulin followed by a short course of oral prednisone, obtaining a full clinical recovery.

Conclusions: Our report confirms previous observations of seronegative autoimmune LE, suggesting the presence of other, still unknown central nervous system antigens representing a target of a postinfectious, autoimmune response in these patients. Moreover, it emphasizes the importance of early recognition and treatment of acute autoimmune LE, to reduce the risk of intensive care unit-related complications and the occurrence of permanent cognitive or behavioral defects.

© 2009 Lippincott Williams & Wilkins, Inc.

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