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AMPA and NMDA receptor antibody autoimmune encephalitis preceded by ocular myasthenia gravis: a case report

BACKGROUND: α-Amino-3-hydroxy-5-methyl-4-isoxazole propionic acid (AMPA) and N-methyl-D-aspartate (NMDA) receptors mediate excitatory neurotransmission in the brain and may be targeted by autoantibodies, leading to autoimmune synaptic encephalitis (AE). AE can be associated with other autoimmune dis...

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Autores principales: Schäfer, Jakob, Christensen, Peter Brøgger, Jensen, Kimmo
Formato: Online Artículo Texto
Lenguaje:English
Publicado: BioMed Central 2023
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9999510/
https://www.ncbi.nlm.nih.gov/pubmed/36899302
http://dx.doi.org/10.1186/s12883-023-03129-2
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author Schäfer, Jakob
Christensen, Peter Brøgger
Jensen, Kimmo
author_facet Schäfer, Jakob
Christensen, Peter Brøgger
Jensen, Kimmo
author_sort Schäfer, Jakob
collection PubMed
description BACKGROUND: α-Amino-3-hydroxy-5-methyl-4-isoxazole propionic acid (AMPA) and N-methyl-D-aspartate (NMDA) receptors mediate excitatory neurotransmission in the brain and may be targeted by autoantibodies, leading to autoimmune synaptic encephalitis (AE). AE can be associated with other autoimmune diseases. However, the cooccurrence of anti-AMPA and NMDA receptor AE together with myasthenia gravis (MG) is unusual. CASE PRESENTATION: A 24-year-old previously healthy male presented with seronegative ocular MG, the diagnosis of which was supported by single-fiber electrophysiology findings. Three months later, he developed AE, initially being positive for AMPA receptor antibodies and subsequently for NMDA receptor antibodies. No underlying malignancy was found. In response to aggressive immunosuppressive treatment, he recovered (modified Rankin Scale (mRS) score change from 5 to 1). Despite some cognitive problems at the 1-year follow-up, which were not revealed using the mRS, he was able to return to his studies. CONCLUSIONS: AE may coexist with other autoimmune disorders. Patients with seronegative MG, including ocular MG, may develop autoimmune encephalitis with more than one cell-surface antibody.
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spelling pubmed-99995102023-03-11 AMPA and NMDA receptor antibody autoimmune encephalitis preceded by ocular myasthenia gravis: a case report Schäfer, Jakob Christensen, Peter Brøgger Jensen, Kimmo BMC Neurol Case Report BACKGROUND: α-Amino-3-hydroxy-5-methyl-4-isoxazole propionic acid (AMPA) and N-methyl-D-aspartate (NMDA) receptors mediate excitatory neurotransmission in the brain and may be targeted by autoantibodies, leading to autoimmune synaptic encephalitis (AE). AE can be associated with other autoimmune diseases. However, the cooccurrence of anti-AMPA and NMDA receptor AE together with myasthenia gravis (MG) is unusual. CASE PRESENTATION: A 24-year-old previously healthy male presented with seronegative ocular MG, the diagnosis of which was supported by single-fiber electrophysiology findings. Three months later, he developed AE, initially being positive for AMPA receptor antibodies and subsequently for NMDA receptor antibodies. No underlying malignancy was found. In response to aggressive immunosuppressive treatment, he recovered (modified Rankin Scale (mRS) score change from 5 to 1). Despite some cognitive problems at the 1-year follow-up, which were not revealed using the mRS, he was able to return to his studies. CONCLUSIONS: AE may coexist with other autoimmune disorders. Patients with seronegative MG, including ocular MG, may develop autoimmune encephalitis with more than one cell-surface antibody. BioMed Central 2023-03-10 /pmc/articles/PMC9999510/ /pubmed/36899302 http://dx.doi.org/10.1186/s12883-023-03129-2 Text en © The Author(s) 2023 https://creativecommons.org/licenses/by/4.0/Open AccessThis article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article's Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article's Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http://creativecommons.org/licenses/by/4.0/ (https://creativecommons.org/licenses/by/4.0/) . The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/ (https://creativecommons.org/publicdomain/zero/1.0/) ) applies to the data made available in this article, unless otherwise stated in a credit line to the data.
spellingShingle Case Report
Schäfer, Jakob
Christensen, Peter Brøgger
Jensen, Kimmo
AMPA and NMDA receptor antibody autoimmune encephalitis preceded by ocular myasthenia gravis: a case report
title AMPA and NMDA receptor antibody autoimmune encephalitis preceded by ocular myasthenia gravis: a case report
title_full AMPA and NMDA receptor antibody autoimmune encephalitis preceded by ocular myasthenia gravis: a case report
title_fullStr AMPA and NMDA receptor antibody autoimmune encephalitis preceded by ocular myasthenia gravis: a case report
title_full_unstemmed AMPA and NMDA receptor antibody autoimmune encephalitis preceded by ocular myasthenia gravis: a case report
title_short AMPA and NMDA receptor antibody autoimmune encephalitis preceded by ocular myasthenia gravis: a case report
title_sort ampa and nmda receptor antibody autoimmune encephalitis preceded by ocular myasthenia gravis: a case report
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9999510/
https://www.ncbi.nlm.nih.gov/pubmed/36899302
http://dx.doi.org/10.1186/s12883-023-03129-2
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