Skip to main content
Top
Published in: BMC Neurology 1/2022

Open Access 01-12-2022 | Herpes Virus | Case report

Caspr2 antibodies in herpes simplex encephalitis: an extension of the spectrum of virus induced autoimmunity? – A case report

Authors: Klaus Berek, Ronny Beer, Astrid Grams, Raimund Helbok, Anna Lindner, Bettina Pfausler, Alois Schiefecker, Florian Deisenhammer, Harald Hegen

Published in: BMC Neurology | Issue 1/2022

Login to get access

Abstract

Background

Post herpes simplex virus (HSV) autoimmune encephalitis has been reported mainly in association with NMDA receptor antibodies, however, never with Caspr2 antibodies.

Case presentation

We report an 82-year old female patient with encephalitis who presented with aphasia, left temporo-mesial hyperintense lesion on MRI, epileptiform discharges on spot electroencephalography, cerebrospinal fluid (CSF) lymphocytic pleocytosis and who showed positive HSV polymerase chain reaction in CSF as well as antibodies against contactin-associated protein-like 2 (Caspr2).

Conclusion

This is the first report of a patient with encephalitis who tested positive for HSV as well as for Caspr2 antibodies.
Literature
1.
2.
go back to reference Venkatesan A, Tunkel AR, Bloch KC, Lauring AS, Sejvar J, Bitnun A, et al. Case definitions, diagnostic algorithms, and priorities in encephalitis: consensus statement of the international encephalitis consortium. Clin Infect Dis. 2013;57(8):1114–28.CrossRef Venkatesan A, Tunkel AR, Bloch KC, Lauring AS, Sejvar J, Bitnun A, et al. Case definitions, diagnostic algorithms, and priorities in encephalitis: consensus statement of the international encephalitis consortium. Clin Infect Dis. 2013;57(8):1114–28.CrossRef
3.
go back to reference Singh TD, Fugate JE, Hocker S, Wijdicks EFM, Aksamit AJ Jr, Rabinstein AA. Predictors of outcome in HSV encephalitis. J Neurol. 2016;263(2):277–89.CrossRef Singh TD, Fugate JE, Hocker S, Wijdicks EFM, Aksamit AJ Jr, Rabinstein AA. Predictors of outcome in HSV encephalitis. J Neurol. 2016;263(2):277–89.CrossRef
4.
go back to reference Armangue T, Leypoldt F, Malaga I, Raspall-Chaure M, Marti I, Nichter C, et al. Herpes simplex virus encephalitis is a trigger of brain autoimmunity. Ann Neurol. 2014;75(2):317–23.CrossRef Armangue T, Leypoldt F, Malaga I, Raspall-Chaure M, Marti I, Nichter C, et al. Herpes simplex virus encephalitis is a trigger of brain autoimmunity. Ann Neurol. 2014;75(2):317–23.CrossRef
5.
go back to reference Armangue T, Spatola M, Vlagea A, Mattozzi S, Carceles-Cordon M, Martinez-Heras E, et al. Frequency, symptoms, risk factors, and outcomes of autoimmune encephalitis after herpes simplex encephalitis: a prospective observational study and retrospective analysis. Lancet Neurol. 2018;17(9):760–72.CrossRef Armangue T, Spatola M, Vlagea A, Mattozzi S, Carceles-Cordon M, Martinez-Heras E, et al. Frequency, symptoms, risk factors, and outcomes of autoimmune encephalitis after herpes simplex encephalitis: a prospective observational study and retrospective analysis. Lancet Neurol. 2018;17(9):760–72.CrossRef
6.
go back to reference Dalmau J, Lancaster E, Martinez-Hernandez E, Rosenfeld MR, Balice-Gordon R. Clinical experience and laboratory investigations in patients with anti-NMDAR encephalitis. Lancet Neurol. 2011;10(1):63–74.CrossRef Dalmau J, Lancaster E, Martinez-Hernandez E, Rosenfeld MR, Balice-Gordon R. Clinical experience and laboratory investigations in patients with anti-NMDAR encephalitis. Lancet Neurol. 2011;10(1):63–74.CrossRef
7.
go back to reference van Sonderen A, Petit-Pedrol M, Dalmau J, Titulaer MJ. The value of LGI1, Caspr2 and voltage-gated potassium channel antibodies in encephalitis. Nat Rev Neurol. 2017;13(5):290–301.CrossRef van Sonderen A, Petit-Pedrol M, Dalmau J, Titulaer MJ. The value of LGI1, Caspr2 and voltage-gated potassium channel antibodies in encephalitis. Nat Rev Neurol. 2017;13(5):290–301.CrossRef
8.
go back to reference Spatola M, Petit-Pedrol M, Simabukuro MM, Armangue T, Castro FJ, Barcelo Artigues MI, et al. Investigations in GABAA receptor antibody-associated encephalitis. Neurology. 2017;88(11):1012–20.CrossRef Spatola M, Petit-Pedrol M, Simabukuro MM, Armangue T, Castro FJ, Barcelo Artigues MI, et al. Investigations in GABAA receptor antibody-associated encephalitis. Neurology. 2017;88(11):1012–20.CrossRef
9.
go back to reference Armangue T, Moris G, Cantarin-Extremera V, Conde CE, Rostasy K, Erro ME, et al. Autoimmune post-herpes simplex encephalitis of adults and teenagers. Neurology. 2015;85(20):1736–43.CrossRef Armangue T, Moris G, Cantarin-Extremera V, Conde CE, Rostasy K, Erro ME, et al. Autoimmune post-herpes simplex encephalitis of adults and teenagers. Neurology. 2015;85(20):1736–43.CrossRef
10.
go back to reference Alexopoulos H, Akrivou S, Mastroyanni S, Antonopoulou M, Dinopoulos A, Giorgi M, et al. Postherpes simplex encephalitis: a case series of viral-triggered autoimmunity, synaptic autoantibodies and response to therapy. Ther Adv Neurol Disord. 2018;11:1756286418768778.CrossRef Alexopoulos H, Akrivou S, Mastroyanni S, Antonopoulou M, Dinopoulos A, Giorgi M, et al. Postherpes simplex encephalitis: a case series of viral-triggered autoimmunity, synaptic autoantibodies and response to therapy. Ther Adv Neurol Disord. 2018;11:1756286418768778.CrossRef
11.
go back to reference Schuster S, Abrante L, Matschke J, Lutgehetmann M, Holst B, Gelderblom M, et al. Fatal PCR-negative herpes simplex virus-1 encephalitis with GABAA receptor antibodies. Neurol Neuroimmunol Neuroinflamm. 2019;6(6):e624.CrossRef Schuster S, Abrante L, Matschke J, Lutgehetmann M, Holst B, Gelderblom M, et al. Fatal PCR-negative herpes simplex virus-1 encephalitis with GABAA receptor antibodies. Neurol Neuroimmunol Neuroinflamm. 2019;6(6):e624.CrossRef
12.
go back to reference Mader S, Gredler V, Schanda K, Rostasy K, Dujmovic I, Pfaller K, et al. Complement activating antibodies to myelin oligodendrocyte glycoprotein in neuromyelitis optica and related disorders. J Neuroinflammation. 2011;8:184.CrossRef Mader S, Gredler V, Schanda K, Rostasy K, Dujmovic I, Pfaller K, et al. Complement activating antibodies to myelin oligodendrocyte glycoprotein in neuromyelitis optica and related disorders. J Neuroinflammation. 2011;8:184.CrossRef
13.
go back to reference Dalmau J, Gleichman AJ, Hughes EG, Rossi JE, Peng X, Lai M, et al. Anti-NMDA-receptor encephalitis: case series and analysis of the effects of antibodies. Lancet Neurol. 2008;7(12):1091–8.CrossRef Dalmau J, Gleichman AJ, Hughes EG, Rossi JE, Peng X, Lai M, et al. Anti-NMDA-receptor encephalitis: case series and analysis of the effects of antibodies. Lancet Neurol. 2008;7(12):1091–8.CrossRef
14.
go back to reference Titulaer MJ, McCracken L, Gabilondo I, Armangue T, Glaser C, Iizuka T, et al. Treatment and prognostic factors for long-term outcome in patients with anti-NMDA receptor encephalitis: an observational cohort study. Lancet Neurol. 2013;12(2):157–65.CrossRef Titulaer MJ, McCracken L, Gabilondo I, Armangue T, Glaser C, Iizuka T, et al. Treatment and prognostic factors for long-term outcome in patients with anti-NMDA receptor encephalitis: an observational cohort study. Lancet Neurol. 2013;12(2):157–65.CrossRef
15.
go back to reference Pruss H, Dalmau J, Harms L, Holtje M, Ahnert-Hilger G, Borowski K, et al. Retrospective analysis of NMDA receptor antibodies in encephalitis of unknown origin. Neurology. 2010;75(19):1735–9.CrossRef Pruss H, Dalmau J, Harms L, Holtje M, Ahnert-Hilger G, Borowski K, et al. Retrospective analysis of NMDA receptor antibodies in encephalitis of unknown origin. Neurology. 2010;75(19):1735–9.CrossRef
16.
go back to reference Wang R, Guan HZ, Ren HT, Wang W, Hong Z, Zhou D. CSF findings in patients with anti-N-methyl-D-aspartate receptor-encephalitis. Seizure. 2015;29:137–42.CrossRef Wang R, Guan HZ, Ren HT, Wang W, Hong Z, Zhou D. CSF findings in patients with anti-N-methyl-D-aspartate receptor-encephalitis. Seizure. 2015;29:137–42.CrossRef
17.
go back to reference van Sonderen A, Thijs RD, Coenders EC, Jiskoot LC, Sanchez E, de Bruijn MA, et al. Anti-LGI1 encephalitis: Clinical syndrome and long-term follow-up. Neurology. 2016;87(14):1449–56.CrossRef van Sonderen A, Thijs RD, Coenders EC, Jiskoot LC, Sanchez E, de Bruijn MA, et al. Anti-LGI1 encephalitis: Clinical syndrome and long-term follow-up. Neurology. 2016;87(14):1449–56.CrossRef
18.
go back to reference Gadoth A, Pittock SJ, Dubey D, McKeon A, Britton JW, Schmeling JE, et al. Expanded phenotypes and outcomes among 256 LGI1/CASPR2-IgG-positive patients. Ann Neurol. 2017;82(1):79–92.CrossRef Gadoth A, Pittock SJ, Dubey D, McKeon A, Britton JW, Schmeling JE, et al. Expanded phenotypes and outcomes among 256 LGI1/CASPR2-IgG-positive patients. Ann Neurol. 2017;82(1):79–92.CrossRef
19.
go back to reference Lin J, Li C, Li A, Liu X, Wang R, Chen C, et al. Encephalitis with antibodies against the GABAB receptor: high mortality and risk factors. Front Neurol. 2019;10:1030.CrossRef Lin J, Li C, Li A, Liu X, Wang R, Chen C, et al. Encephalitis with antibodies against the GABAB receptor: high mortality and risk factors. Front Neurol. 2019;10:1030.CrossRef
20.
go back to reference Laurido-Soto O, Brier MR, Simon LE, McCullough A, Bucelli RC, Day GS. Patient characteristics and outcome associations in AMPA receptor encephalitis. J Neurol. 2019;266(2):450–60.CrossRef Laurido-Soto O, Brier MR, Simon LE, McCullough A, Bucelli RC, Day GS. Patient characteristics and outcome associations in AMPA receptor encephalitis. J Neurol. 2019;266(2):450–60.CrossRef
21.
go back to reference Jarius S, Paul F, Franciotta D, Ruprecht K, Ringelstein M, Bergamaschi R, et al. Cerebrospinal fluid findings in aquaporin-4 antibody positive neuromyelitis optica: results from 211 lumbar punctures. J Neurol Sci. 2011;306(1–2):82–90.CrossRef Jarius S, Paul F, Franciotta D, Ruprecht K, Ringelstein M, Bergamaschi R, et al. Cerebrospinal fluid findings in aquaporin-4 antibody positive neuromyelitis optica: results from 211 lumbar punctures. J Neurol Sci. 2011;306(1–2):82–90.CrossRef
22.
go back to reference Wingerchuk DM, Banwell B, Bennett JL, Cabre P, Carroll W, Chitnis T, et al. International consensus diagnostic criteria for neuromyelitis optica spectrum disorders. Neurology. 2015;85(2):177–89.CrossRef Wingerchuk DM, Banwell B, Bennett JL, Cabre P, Carroll W, Chitnis T, et al. International consensus diagnostic criteria for neuromyelitis optica spectrum disorders. Neurology. 2015;85(2):177–89.CrossRef
23.
go back to reference Hegen H, Reindl M. Recent developments in MOG-IgG associated neurological disorders. Ther Adv Neurol Disord. 2020;13:1756286420945135.CrossRef Hegen H, Reindl M. Recent developments in MOG-IgG associated neurological disorders. Ther Adv Neurol Disord. 2020;13:1756286420945135.CrossRef
24.
go back to reference Joubert B, Saint-Martin M, Noraz N, Picard G, Rogemond V, Ducray F, et al. Characterization of a subtype of autoimmune encephalitis with anti-contactin-associated protein-like 2 antibodies in the cerebrospinal fluid, prominent limbic symptoms, and seizures. JAMA Neurol. 2016;73(9):1115–24.CrossRef Joubert B, Saint-Martin M, Noraz N, Picard G, Rogemond V, Ducray F, et al. Characterization of a subtype of autoimmune encephalitis with anti-contactin-associated protein-like 2 antibodies in the cerebrospinal fluid, prominent limbic symptoms, and seizures. JAMA Neurol. 2016;73(9):1115–24.CrossRef
25.
go back to reference Bien CG, Mirzadjanova Z, Baumgartner C, Onugoren MD, Grunwald T, Holtkamp M, et al. Anti-contactin-associated protein-2 encephalitis: relevance of antibody titres, presentation and outcome. Eur J Neurol. 2017;24(1):175–86.CrossRef Bien CG, Mirzadjanova Z, Baumgartner C, Onugoren MD, Grunwald T, Holtkamp M, et al. Anti-contactin-associated protein-2 encephalitis: relevance of antibody titres, presentation and outcome. Eur J Neurol. 2017;24(1):175–86.CrossRef
26.
go back to reference McCracken L, Zhang J, Greene M, Crivaro A, Gonzalez J, Kamoun M, et al. Improving the antibody-based evaluation of autoimmune encephalitis. Neurol Neuroimmunol Neuroinflamm. 2017;4(6):e404.CrossRef McCracken L, Zhang J, Greene M, Crivaro A, Gonzalez J, Kamoun M, et al. Improving the antibody-based evaluation of autoimmune encephalitis. Neurol Neuroimmunol Neuroinflamm. 2017;4(6):e404.CrossRef
27.
go back to reference Dahm L, Ott C, Steiner J, Stepniak B, Teegen B, Saschenbrecker S, et al. Seroprevalence of autoantibodies against brain antigens in health and disease. Ann Neurol. 2014;76(1):82–94.CrossRef Dahm L, Ott C, Steiner J, Stepniak B, Teegen B, Saschenbrecker S, et al. Seroprevalence of autoantibodies against brain antigens in health and disease. Ann Neurol. 2014;76(1):82–94.CrossRef
Metadata
Title
Caspr2 antibodies in herpes simplex encephalitis: an extension of the spectrum of virus induced autoimmunity? – A case report
Authors
Klaus Berek
Ronny Beer
Astrid Grams
Raimund Helbok
Anna Lindner
Bettina Pfausler
Alois Schiefecker
Florian Deisenhammer
Harald Hegen
Publication date
01-12-2022
Publisher
BioMed Central
Published in
BMC Neurology / Issue 1/2022
Electronic ISSN: 1471-2377
DOI
https://doi.org/10.1186/s12883-022-02637-x

Other articles of this Issue 1/2022

BMC Neurology 1/2022 Go to the issue