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Intravenous Immunoglobulin Treatment Did Not Improve Tics in a Patient With Gilles de la Tourette Syndrome and Intrathecal Antibody Synthesis

Gilles de la Tourette syndrome (GTS) is a neuropsychiatric disorder characterized by motor and vocal tics. There are several hypotheses as to the cause of the disease. One of which suggests that the immune system is involved in the pathophysiology of GTS. Here, we present a 40-year-old female patien...

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Autores principales: Szejko, Natalia, Fremer, Carolin, Sühs, Kurt-Wolfram, Macul Ferreira de Barros, Pedro, Müller-Vahl, Kirsten R.
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Frontiers Media S.A. 2020
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7059101/
https://www.ncbi.nlm.nih.gov/pubmed/32180755
http://dx.doi.org/10.3389/fneur.2020.00110
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author Szejko, Natalia
Fremer, Carolin
Sühs, Kurt-Wolfram
Macul Ferreira de Barros, Pedro
Müller-Vahl, Kirsten R.
author_facet Szejko, Natalia
Fremer, Carolin
Sühs, Kurt-Wolfram
Macul Ferreira de Barros, Pedro
Müller-Vahl, Kirsten R.
author_sort Szejko, Natalia
collection PubMed
description Gilles de la Tourette syndrome (GTS) is a neuropsychiatric disorder characterized by motor and vocal tics. There are several hypotheses as to the cause of the disease. One of which suggests that the immune system is involved in the pathophysiology of GTS. Here, we present a 40-year-old female patient with a typical history and clinical presentation of GTS with tics and psychiatric comorbidities, in whom positive oligoclonal bands (OCBs) type 2 in cerebral spinal fluid (CSF) were detected in an earlier study. At that time point, all other investigations were unremarkable (including neurological examination and cMRI), but 2 years later, she presented with further neurological symptoms including tetraparesis mostly affecting the left limbs, distal hypesthesia and paresthesia, and dyspnea. Since further examinations (including EMG, MRI, CSF, virological, and bacteriological tests, as well as autoimmune-encephalitis antibodies) revealed normal results, based on clinical presentation, the diagnosis of Guillain-Barré-like immune-mediated neuropathy was made and treatment with intravenous immunoglobulins (IVIg) (30 g/day for 5 days) was initiated resulting in complete remission of immune-mediated neuropathy. Based on the “immune hypothesis” of GTS, we were interested in whether in this patient positive CSF OCBs might serve as a biomarker for treatment response of tics and additional GTS-related psychiatric symptoms to IVIg, and therefore assessed tics, premonitory urges, and psychiatric comorbidities before and several times after the IVIg treatment. However, even though immune-mediated neuropathy resolved after treatment, tics, premonitory urges, and comorbidities remained unchanged. Thus, this case study suggests that treatment with IVIg is not effective in the treatment of GTS—even in a patient with intrathecal antibody synthesis as expressed by CSF isolated OCBs.
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spelling pubmed-70591012020-03-16 Intravenous Immunoglobulin Treatment Did Not Improve Tics in a Patient With Gilles de la Tourette Syndrome and Intrathecal Antibody Synthesis Szejko, Natalia Fremer, Carolin Sühs, Kurt-Wolfram Macul Ferreira de Barros, Pedro Müller-Vahl, Kirsten R. Front Neurol Neurology Gilles de la Tourette syndrome (GTS) is a neuropsychiatric disorder characterized by motor and vocal tics. There are several hypotheses as to the cause of the disease. One of which suggests that the immune system is involved in the pathophysiology of GTS. Here, we present a 40-year-old female patient with a typical history and clinical presentation of GTS with tics and psychiatric comorbidities, in whom positive oligoclonal bands (OCBs) type 2 in cerebral spinal fluid (CSF) were detected in an earlier study. At that time point, all other investigations were unremarkable (including neurological examination and cMRI), but 2 years later, she presented with further neurological symptoms including tetraparesis mostly affecting the left limbs, distal hypesthesia and paresthesia, and dyspnea. Since further examinations (including EMG, MRI, CSF, virological, and bacteriological tests, as well as autoimmune-encephalitis antibodies) revealed normal results, based on clinical presentation, the diagnosis of Guillain-Barré-like immune-mediated neuropathy was made and treatment with intravenous immunoglobulins (IVIg) (30 g/day for 5 days) was initiated resulting in complete remission of immune-mediated neuropathy. Based on the “immune hypothesis” of GTS, we were interested in whether in this patient positive CSF OCBs might serve as a biomarker for treatment response of tics and additional GTS-related psychiatric symptoms to IVIg, and therefore assessed tics, premonitory urges, and psychiatric comorbidities before and several times after the IVIg treatment. However, even though immune-mediated neuropathy resolved after treatment, tics, premonitory urges, and comorbidities remained unchanged. Thus, this case study suggests that treatment with IVIg is not effective in the treatment of GTS—even in a patient with intrathecal antibody synthesis as expressed by CSF isolated OCBs. Frontiers Media S.A. 2020-02-28 /pmc/articles/PMC7059101/ /pubmed/32180755 http://dx.doi.org/10.3389/fneur.2020.00110 Text en Copyright © 2020 Szejko, Fremer, Sühs, Macul Ferreira de Barros and Müller-Vahl. http://creativecommons.org/licenses/by/4.0/ This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY). The use, distribution or reproduction in other forums is permitted, provided the original author(s) and the copyright owner(s) are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms.
spellingShingle Neurology
Szejko, Natalia
Fremer, Carolin
Sühs, Kurt-Wolfram
Macul Ferreira de Barros, Pedro
Müller-Vahl, Kirsten R.
Intravenous Immunoglobulin Treatment Did Not Improve Tics in a Patient With Gilles de la Tourette Syndrome and Intrathecal Antibody Synthesis
title Intravenous Immunoglobulin Treatment Did Not Improve Tics in a Patient With Gilles de la Tourette Syndrome and Intrathecal Antibody Synthesis
title_full Intravenous Immunoglobulin Treatment Did Not Improve Tics in a Patient With Gilles de la Tourette Syndrome and Intrathecal Antibody Synthesis
title_fullStr Intravenous Immunoglobulin Treatment Did Not Improve Tics in a Patient With Gilles de la Tourette Syndrome and Intrathecal Antibody Synthesis
title_full_unstemmed Intravenous Immunoglobulin Treatment Did Not Improve Tics in a Patient With Gilles de la Tourette Syndrome and Intrathecal Antibody Synthesis
title_short Intravenous Immunoglobulin Treatment Did Not Improve Tics in a Patient With Gilles de la Tourette Syndrome and Intrathecal Antibody Synthesis
title_sort intravenous immunoglobulin treatment did not improve tics in a patient with gilles de la tourette syndrome and intrathecal antibody synthesis
topic Neurology
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7059101/
https://www.ncbi.nlm.nih.gov/pubmed/32180755
http://dx.doi.org/10.3389/fneur.2020.00110
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