Efficacy of Tocilizumab in Limbic Encephalitis with Anti-CASPR2 Antibodies

We report the case of a 64-year-old man who presented with subacute memory, balance impairment, behavioral and mood changes, and epileptic seizures. Magnetic resonance imaging (MRI) showed bilateral hippocampal abnormalities. Brain [18F]-FDG fluorodeoxyglucose positron emission tomography (PET) reve...

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Main Authors: Maurizio Benucci, Luciana Tramacere, Maria Infantino, Mariangela Manfredi, Valentina Grossi, Arianna Damiani, Francesca Li Gobbi, Maristella Piccininni, Gaetano Zaccara, Massimo Cincotta
Format: Article
Language:English
Published: Hindawi Limited 2020-01-01
Series:Case Reports in Neurological Medicine
Online Access:http://dx.doi.org/10.1155/2020/5697670
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spelling doaj-5604d688e6004e75bc8512c53c8910472020-11-25T01:15:00ZengHindawi LimitedCase Reports in Neurological Medicine2090-66682090-66762020-01-01202010.1155/2020/56976705697670Efficacy of Tocilizumab in Limbic Encephalitis with Anti-CASPR2 AntibodiesMaurizio Benucci0Luciana Tramacere1Maria Infantino2Mariangela Manfredi3Valentina Grossi4Arianna Damiani5Francesca Li Gobbi6Maristella Piccininni7Gaetano Zaccara8Massimo Cincotta9Rheumatology Unit, S. Giovanni di Dio Hospital, Florence, ItalyNeurology Unit, S. Giovanni di Dio Hospital, Florence, ItalyImmunology and Allergology Laboratory, S. Giovanni di Dio Hospital, Florence, ItalyImmunology and Allergology Laboratory, S. Giovanni di Dio Hospital, Florence, ItalyImmunology and Allergology Laboratory, S. Giovanni di Dio Hospital, Florence, ItalyRheumatology Unit, S. Giovanni di Dio Hospital, Florence, ItalyRheumatology Unit, S. Giovanni di Dio Hospital, Florence, ItalyNeurology Unit, S. Giovanni di Dio Hospital, Florence, ItalyNeurology Unit, S. Giovanni di Dio Hospital, Florence, ItalyNeurology Unit, S. Giovanni di Dio Hospital, Florence, ItalyWe report the case of a 64-year-old man who presented with subacute memory, balance impairment, behavioral and mood changes, and epileptic seizures. Magnetic resonance imaging (MRI) showed bilateral hippocampal abnormalities. Brain [18F]-FDG fluorodeoxyglucose positron emission tomography (PET) revealed hypometabolism in both the temporal lobe as well as in the left insular and parietal regions. The clinical and neuroradiological picture and the detection of anti-CASPR2 antibodies in serum oriented the diagnosis towards autoimmune limbic encephalitis. Intravenous high-dose steroid and immunoglobulin treatments were ineffective. We did not use rituximab for the presence of antibodies to HbcAg positivity. Tocilizumab given intravenously 8 mg/kg once a month for six months and then subcutaneously 162 mg every week for six months resulted in clinical and neuroradiological improvement. These data support the efficacy of tocilizumab in autoimmune limbic encephalitis associated with anti-CASPR2 antibodies, which has been sporadically reported in the literature.http://dx.doi.org/10.1155/2020/5697670
collection DOAJ
language English
format Article
sources DOAJ
author Maurizio Benucci
Luciana Tramacere
Maria Infantino
Mariangela Manfredi
Valentina Grossi
Arianna Damiani
Francesca Li Gobbi
Maristella Piccininni
Gaetano Zaccara
Massimo Cincotta
spellingShingle Maurizio Benucci
Luciana Tramacere
Maria Infantino
Mariangela Manfredi
Valentina Grossi
Arianna Damiani
Francesca Li Gobbi
Maristella Piccininni
Gaetano Zaccara
Massimo Cincotta
Efficacy of Tocilizumab in Limbic Encephalitis with Anti-CASPR2 Antibodies
Case Reports in Neurological Medicine
author_facet Maurizio Benucci
Luciana Tramacere
Maria Infantino
Mariangela Manfredi
Valentina Grossi
Arianna Damiani
Francesca Li Gobbi
Maristella Piccininni
Gaetano Zaccara
Massimo Cincotta
author_sort Maurizio Benucci
title Efficacy of Tocilizumab in Limbic Encephalitis with Anti-CASPR2 Antibodies
title_short Efficacy of Tocilizumab in Limbic Encephalitis with Anti-CASPR2 Antibodies
title_full Efficacy of Tocilizumab in Limbic Encephalitis with Anti-CASPR2 Antibodies
title_fullStr Efficacy of Tocilizumab in Limbic Encephalitis with Anti-CASPR2 Antibodies
title_full_unstemmed Efficacy of Tocilizumab in Limbic Encephalitis with Anti-CASPR2 Antibodies
title_sort efficacy of tocilizumab in limbic encephalitis with anti-caspr2 antibodies
publisher Hindawi Limited
series Case Reports in Neurological Medicine
issn 2090-6668
2090-6676
publishDate 2020-01-01
description We report the case of a 64-year-old man who presented with subacute memory, balance impairment, behavioral and mood changes, and epileptic seizures. Magnetic resonance imaging (MRI) showed bilateral hippocampal abnormalities. Brain [18F]-FDG fluorodeoxyglucose positron emission tomography (PET) revealed hypometabolism in both the temporal lobe as well as in the left insular and parietal regions. The clinical and neuroradiological picture and the detection of anti-CASPR2 antibodies in serum oriented the diagnosis towards autoimmune limbic encephalitis. Intravenous high-dose steroid and immunoglobulin treatments were ineffective. We did not use rituximab for the presence of antibodies to HbcAg positivity. Tocilizumab given intravenously 8 mg/kg once a month for six months and then subcutaneously 162 mg every week for six months resulted in clinical and neuroradiological improvement. These data support the efficacy of tocilizumab in autoimmune limbic encephalitis associated with anti-CASPR2 antibodies, which has been sporadically reported in the literature.
url http://dx.doi.org/10.1155/2020/5697670
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