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Published in: BMC Neurology 1/2023

Open Access 01-12-2023 | Myasthenia Gravis | Case report

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

Authors: Jakob Schäfer, Peter Brøgger Christensen, Kimmo Jensen

Published in: BMC Neurology | Issue 1/2023

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Abstract

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.
Literature
1.
2.
go back to reference Lai M, Hughes EG, Peng X, Zhou L, Gleichman AJ, Shu H, et al. AMPA receptor antibodies in limbic encephalitis alter synaptic receptor location. Ann Neurol. 2009;65:424–34.CrossRefPubMedPubMedCentral Lai M, Hughes EG, Peng X, Zhou L, Gleichman AJ, Shu H, et al. AMPA receptor antibodies in limbic encephalitis alter synaptic receptor location. Ann Neurol. 2009;65:424–34.CrossRefPubMedPubMedCentral
3.
go back to reference Martinez-Hernandez E, Guasp M, García-Serra A, Maudes E, Ariño H, Sepulveda M, et al. Clinical significance of anti-NMDAR concurrent with glial or neuronal surface antibodies. Neurology. 2020;94:e2302–10.CrossRefPubMed Martinez-Hernandez E, Guasp M, García-Serra A, Maudes E, Ariño H, Sepulveda M, et al. Clinical significance of anti-NMDAR concurrent with glial or neuronal surface antibodies. Neurology. 2020;94:e2302–10.CrossRefPubMed
4.
go back to reference Linnoila JJ, Binnicker MJ, Majed M, McKeon A. CSF herpes virus and autoantibody profiles in the evaluation of encephalitis. Neurol Neuroimmunol Neuroinflamm. 2016;3(4):e245.CrossRefPubMedPubMedCentral Linnoila JJ, Binnicker MJ, Majed M, McKeon A. CSF herpes virus and autoantibody profiles in the evaluation of encephalitis. Neurol Neuroimmunol Neuroinflamm. 2016;3(4):e245.CrossRefPubMedPubMedCentral
6.
go back to reference Titulaer MJ, McCrachen L, Gabilondo I, Armanqué T, Glaser C, Iizuka T, et al. Treatment and prognostic factors for long-term outcome in patients with anti-N-Methy-D-aspartate (NMDA) receptor encephalitis: a cohort study. Lancet Neurol. 2013;12:157–65.CrossRefPubMedPubMedCentral Titulaer MJ, McCrachen L, Gabilondo I, Armanqué T, Glaser C, Iizuka T, et al. Treatment and prognostic factors for long-term outcome in patients with anti-N-Methy-D-aspartate (NMDA) receptor encephalitis: a cohort study. Lancet Neurol. 2013;12:157–65.CrossRefPubMedPubMedCentral
7.
go back to reference Dalmau J, Gleichmann AJ, Hughes EG, Rossi JE, Peng X, Lai M, et al. Anti-NMDA-receptor encephalitis. Case series and analysis of the effecs of antibodies. Lancet Neurol. 2008;7(12):1091–8.CrossRefPubMedPubMedCentral Dalmau J, Gleichmann AJ, Hughes EG, Rossi JE, Peng X, Lai M, et al. Anti-NMDA-receptor encephalitis. Case series and analysis of the effecs of antibodies. Lancet Neurol. 2008;7(12):1091–8.CrossRefPubMedPubMedCentral
8.
go back to reference Höftberger R, van Sonderen A, Leypoldt F, Houghton D, Geschwind M, Gelfand J, et al. Encephalitis and AMPA receptor antibodies: novel findings in a case series of 22 patients. Neurology. 2015;84:2403–12.CrossRefPubMedPubMedCentral Höftberger R, van Sonderen A, Leypoldt F, Houghton D, Geschwind M, Gelfand J, et al. Encephalitis and AMPA receptor antibodies: novel findings in a case series of 22 patients. Neurology. 2015;84:2403–12.CrossRefPubMedPubMedCentral
11.
15.
go back to reference Finke C, Kopp UA, Prüss H, Dalmau J, Wandinger K-P, Ploner CJ. Cognitive deficits following anti-NMDA receptor encephalitis. J Neurol Neurosurg Psychiatry. 2012;83:195–8.CrossRefPubMed Finke C, Kopp UA, Prüss H, Dalmau J, Wandinger K-P, Ploner CJ. Cognitive deficits following anti-NMDA receptor encephalitis. J Neurol Neurosurg Psychiatry. 2012;83:195–8.CrossRefPubMed
17.
go back to reference Zhao J, Wang C, Xu X, Zhang Y, Ren H, Ren Z, et al. Coexistence of autoimmune encephalitis and other systemic autoimmune diseases. Front Neurol. 2019;10:11–42.CrossRef Zhao J, Wang C, Xu X, Zhang Y, Ren H, Ren Z, et al. Coexistence of autoimmune encephalitis and other systemic autoimmune diseases. Front Neurol. 2019;10:11–42.CrossRef
Metadata
Title
AMPA and NMDA receptor antibody autoimmune encephalitis preceded by ocular myasthenia gravis: a case report
Authors
Jakob Schäfer
Peter Brøgger Christensen
Kimmo Jensen
Publication date
01-12-2023
Publisher
BioMed Central
Published in
BMC Neurology / Issue 1/2023
Electronic ISSN: 1471-2377
DOI
https://doi.org/10.1186/s12883-023-03129-2

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