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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 [(18)F]-FDG fluorodeoxyglucose positron emission tomography (PET) re...

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Autores principales: Benucci, Maurizio, Tramacere, Luciana, Infantino, Maria, Manfredi, Mariangela, Grossi, Valentina, Damiani, Arianna, Gobbi, Francesca Li, Piccininni, Maristella, Zaccara, Gaetano, Cincotta, Massimo
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Hindawi 2020
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7042499/
https://www.ncbi.nlm.nih.gov/pubmed/32110453
http://dx.doi.org/10.1155/2020/5697670
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author Benucci, Maurizio
Tramacere, Luciana
Infantino, Maria
Manfredi, Mariangela
Grossi, Valentina
Damiani, Arianna
Gobbi, Francesca Li
Piccininni, Maristella
Zaccara, Gaetano
Cincotta, Massimo
author_facet Benucci, Maurizio
Tramacere, Luciana
Infantino, Maria
Manfredi, Mariangela
Grossi, Valentina
Damiani, Arianna
Gobbi, Francesca Li
Piccininni, Maristella
Zaccara, Gaetano
Cincotta, Massimo
author_sort Benucci, Maurizio
collection PubMed
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 [(18)F]-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.
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spelling pubmed-70424992020-02-27 Efficacy of Tocilizumab in Limbic Encephalitis with Anti-CASPR2 Antibodies Benucci, Maurizio Tramacere, Luciana Infantino, Maria Manfredi, Mariangela Grossi, Valentina Damiani, Arianna Gobbi, Francesca Li Piccininni, Maristella Zaccara, Gaetano Cincotta, Massimo Case Rep Neurol Med Case Report 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 [(18)F]-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. Hindawi 2020-02-14 /pmc/articles/PMC7042499/ /pubmed/32110453 http://dx.doi.org/10.1155/2020/5697670 Text en Copyright © 2020 Maurizio Benucci et al. http://creativecommons.org/licenses/by/4.0/ This is an open access article distributed under the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.
spellingShingle Case Report
Benucci, Maurizio
Tramacere, Luciana
Infantino, Maria
Manfredi, Mariangela
Grossi, Valentina
Damiani, Arianna
Gobbi, Francesca Li
Piccininni, Maristella
Zaccara, Gaetano
Cincotta, Massimo
Efficacy of Tocilizumab in Limbic Encephalitis with Anti-CASPR2 Antibodies
title 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_short Efficacy of Tocilizumab in Limbic Encephalitis with Anti-CASPR2 Antibodies
title_sort efficacy of tocilizumab in limbic encephalitis with anti-caspr2 antibodies
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7042499/
https://www.ncbi.nlm.nih.gov/pubmed/32110453
http://dx.doi.org/10.1155/2020/5697670
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