Seronegative autoimmune encephalitis associated with COVID-19: case report

Authors

  • Miguel A Vences Universidad César Vallejo, Escuela de Medicina. Piura, Perú. https://orcid.org/0000-0002-8538-6242
  • Maria Fe Albujar Hospital Nacional Edgardo Rebagliati Martins, Departamento de Neurología. Lima, Perú.
  • Marcela Araujo Hospital Nacional Edgardo Rebagliati Martins, Departamento de Neurología. Lima, Perú. https://orcid.org/0000-0003-4910-3484
  • Diego Canales Hospital Nacional Edgardo Rebagliati Martins, Departamento de Neurología. Lima, Perú.
  • Edu Cardenas Hospital Nacional Edgardo Rebagliati Martins, Departamento de Neurología. Lima, Perú. https://orcid.org/0000-0002-5654-0781
  • Ebelin Barja Hospital Nacional Edgardo Rebagliati Martins, Departamento de Neurología. Lima, Perú.
  • Arturo Alvarez Hospital Nacional Edgardo Rebagliati Martins, Departamento de Neurología. Lima, Perú.
  • Diego Urrunaga-Pastor Universidad Científica del Sur, Facultad de Ciencias de la Salud. Lima, Perú. https://orcid.org/0000-0002-8339-162X

DOI:

https://doi.org/10.20453/rnp.v87i2.5254

Keywords:

autoimmune encephalitis, coronavirus disease-2019 (COVID-19), neurological manifestation, immune therapy, antibody

Abstract

Autoimmune encephalitis has been reported to be temporarily associated with different previous viral infections, but to date its association with COVID-19 has been rarely described. The case of a 47-year-old, self-sufficient woman with no history of COVID-19 vaccination, with respiratory symptoms and a positive antigen test for SARS CoV-2 infection, is reported. She was initially seen at the ER of a local hospital, with a history of heteroaggressiveness, akinetic mutism, epileptic crisis, and hyperreflexia. Negative infectious, autoimmune and neoplastic tests. The autoimmune encephalitis panel in CSF was negative. We administered empirical treatment with methylprednisolone and immunoglobulin, without a favorable therapeutic response; so treatment with cyclophosphamide was initiated with a favorable clinical evolution, but with severe residual neurocognitive manifestations. The fact that the initial clinical presentation was severe and unresponsive to first-line treatment despite being timely employed, could indicate a greater probability of long-term sequelae.

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Author Biographies

Miguel A Vences, Universidad César Vallejo, Escuela de Medicina. Piura, Perú.

   

Maria Fe Albujar, Hospital Nacional Edgardo Rebagliati Martins, Departamento de Neurología. Lima, Perú.

   

Marcela Araujo, Hospital Nacional Edgardo Rebagliati Martins, Departamento de Neurología. Lima, Perú.

  

Diego Canales, Hospital Nacional Edgardo Rebagliati Martins, Departamento de Neurología. Lima, Perú.

    

Edu Cardenas, Hospital Nacional Edgardo Rebagliati Martins, Departamento de Neurología. Lima, Perú.

   

Ebelin Barja, Hospital Nacional Edgardo Rebagliati Martins, Departamento de Neurología. Lima, Perú.

   

Arturo Alvarez, Hospital Nacional Edgardo Rebagliati Martins, Departamento de Neurología. Lima, Perú.

   

Diego Urrunaga-Pastor, Universidad Científica del Sur, Facultad de Ciencias de la Salud. Lima, Perú.

   

References

World Health Organization. WHO Coronavirus (COVID-19) Dashboard [Internet]. Available from: https://covid19.who.int/table

Misra S, Kolappa K, Prasad M, Radhakrishnan D, Thakur KT, Solomon T, et al. Frequency of neurologic manifestations in COVID-19: a systematic review and meta-analysis. Neurology [Internet]. 2021; 97(23): e2269-e2281. Available from: https://doi.org/10.1212/WNL.0000000000012930

Burr T, Barton C, Doll E, Lakhotia A, Sweeney M. N-methyl-d-aspartate receptor encephalitis associated with COVID-19 infection in a toddler. Pediatr Neurol [Internet]. 2021; 114: 75-76. Available from: https://doi.org/10.1016/j.pediatrneurol.2020.10.002

Abboud H, Probasco JC, Irani S, Ances B, Benavides DR, Bradshaw M, et al. Autoimmune encephalitis: proposed best practice recommendations for diagnosis and acute management. J Neurol Neurosurg Psychiatry [Internet]. 2021; 92(7): 757-768. Available from: https://doi.org/10.1136/jnnp-2020-325300

Dubey D, Pittock SJ, Kelly CR, McKeon A, Lopez-Chiriboga AS, Lennon VA, et al. Autoimmune encephalitis epidemiology and a comparison to infectious encephalitis. Ann Neurol [Internet]. 2018; 83(1): 166-177. Available from: https://doi.org/10.1002/ana.25131

Vences MA, Saquisela VV, Barreto-Acevedo E, Zuñiga MA. Encefalitis anti-NMDAR Encephalitis: reporte de caso con seguimiento a largo plazo. Rev Neuropsiquiatr [Internet]. 2020; 83(2): 110-115. Available from: http://dx.doi.org/10.20453/rnp.v83i2.3753

Morishima T, Togashi T, Yokota S, Okuno Y, Miyazaki C, Tashiro M, et al. Encephalitis and encephalopathy associated with an influenza epidemic in Japan. Clin Infect Dis [Internet]. 2002; 35(5): 512-517. Available from: https://doi.org/10.1086/341407

Zuhorn F, Graf T, Klingebiel R, Schäbitz WR, Rogalewski A. Postvaccinal encephalitis after ChAdOx1 nCov-19. Ann Neurol [Internet]. 2021; 90(3): 506-511. Available from: https://doi.org/10.1002/ana.26182

Pilotto A, Masciocchi S, Volonghi I, Crabbio M, Magni E, De Giuli V, et al. Clinical presentation and outcomes of severe acute respiratory syndrome coronavirus 2–related encephalitis: the ENCOVID multicenter study. J Infect Dis. 2021; 223(1): 28-37. Available from: https://doi.org/10.1093/infdis/jiaa609

Sanchez CV, Theel E, Binnicker M, Toledano M, McKeon A. Autoimmune encephalitis after SARS-CoV-2 infection: case frequency, findings, and outcomes. Neurology [Internet]. 2021; 97(23): e2262-e2268. Available from: https://doi.org/10.1212/wnl.0000000000012931

Palacio-Toro MA, Hernández-Botero JS, Duque-Montoya D, Osorio Y, Echeverry A, Osorio-Maldonado JJ, et al. Acute meningoencephalitis associated with SARS-CoV-2 infection in Colombia. J Neurovirol [Internet]. 2021; 27: 960-965. Available from: https://doi.org/10.1007/s13365-021-01023-6

Zanin L, Saraceno G, Renisi G, Signorini L, Battaglia L, Ferrara M, et al. Delayed onset of fatal encephalitis in a COVID-19 positive patient. Int J Neurosci [Internet]. 2021; 133(1): 77-80. Available from: https://doi.org/10.1080/00207454.2021.1886095

Durovic E, Bien C, Bien CG, Isenmann S. MOG antibody-associated encephalitis secondary to Covid-19: case report. BMC Neurol [Internet]. 2021; 21: 414. Available from: https://doi.org/10.1186/s12883-021-02449-5

Zandifar A, Badrfam R. COVID-19 and anti-N-methyl-d-aspartate receptor (anti-NMDAR) encephalitis: are we facing an increase in the prevalence of autoimmune encephalitis? J Med Virol [Internet]. 2021; 93(4): 1913-1914. Available from: https://doi.org/10.1002/jmv.26745

Valadez-Calderon J, Ordinola A, Rodriguez-Chavez E, Vera-Lastra O. Co-expression of anti-NMDAR and anti-GAD65 antibodies. A case of autoimmune encephalitis in a post-COVID-19 patient. Neurología [Internet]. 2021; 37(6): 503-504. Available from: https://doi.org/10.1016/j.nrl.2021.09.003

Nabizadeh F, Balabandian M, Sodeifian F, Rezaei N, Rostami MR, Naser Moghadasi A. Autoimmune encephalitis associated with COVID-19: a systematic review. Mult Scler Relat Disord [Internet]. 2022; 62: 103795. Available from: https://doi.org/10.1016/j.msard.2022.103795

Wang M, Li T, Qiao F, Wang L, Li C, Gong Y. Coronavirus disease 2019 associated with aggressive neurological and mental abnormalities confirmed based on cerebrospinal fluid antibodies: a case report. Medicine [Internet]. 2020; 99(36): e21428. Available from: https://doi.org/10.1097/md.0000000000021428

Khoo A, McLoughlin B, Cheema S, Weil RS, Lambert C, Manji H, et al. Postinfectious brainstem encephalitis associated with SARS-CoV-2. J Neurol Neurosurg Psychiatry [Internet]. 2020; 91(9): 1013-1014. Available from: https://doi.org/10.1136/jnnp-2020-323816

Grimaldi S, Lagarde S, Harlé JR, Boucraut J, Guedj E. Autoimmune encephalitis concomitant with SARS-CoV-2 infection: insight from 18F-FDG PET imaging and neuronal autoantibodies. J Nucl Med [Internet]. 2020; 61(12): 1726-1729. Available from: https://doi.org/10.2967/jnumed.120.249292

Ayatollahi P, Tarazi A, Wennberg R. Possible autoimmune encephalitis with claustrum sign in case of acute SARS-CoV-2 infection. Can J Neurol Sci [Internet]. 2021; 48(3): 430-432. Available from: https://doi.org/10.1017/cjn.2020.209

Samim MM, Dhar D, Goyal S, Dey T, Parvin N, Shah RD, et al. AI-CoV study: autoimmune encephalitis associated with COVID-19 and its vaccines—A systematic review. J Clin Neurol [Internet]. 2022; 18(6): 692-710. Available from: https://doi.org/10.3988/jcn.2022.18.6.692

Bordonne M, Chawki MB, Doyen M, Kas A, Guedj E, Tyvaert L, et al. Brain 18F-FDG PET for the diagnosis of autoimmune encephalitis: a systematic review and a meta-analysis. Eur J Nucl Med Mol Imaging [Internet]. 2021; 48(12): 3847-3858. Available from: https://doi.org/10.1007/s00259-021-05299-y

Uy CE, Binks S, Irani SR. Autoimmune encephalitis: clinical spectrum and management. Pract Neurol [Internet]. 2021; 21(5): 412-423. Available from: https://doi.org/10.1136/practneurol-2020-002567

Vasconcelos G, Barreira R, Antoniollo KE, Pinheiro AM, Maia CF, Alves DM, et al. Autoimmune encephalitis in Latin America: a critical review. Front Neurol [Internet]. 2021; 11: 1846. Available from: https://doi.org/10.3389/fneur.2020.606350

Balu R, McCracken L, Lancaster E, Graus F, Dalmau J, Titulaer MJ. A score that predicts 1-year functional status in patients with anti-NMDA receptor encephalitis. Neurology [Internet]. 2019; 92(3): e244-e252. Available from: https://doi.org/10.1212/wnl.0000000000006783

Titulaer MJ, McCracken L, Gabilondo I, Armangué 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 [Internet]. 2013; 12(2): 157-165. Available from: https://doi.org/10.1016/s1474-4422(12)70310-1

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Published

2024-06-28 — Updated on 2024-07-13

How to Cite

1.
Vences MA, Albujar MF, Araujo M, Canales D, Cardenas E, Barja E, et al. Seronegative autoimmune encephalitis associated with COVID-19: case report . Rev Neuropsiquiatr [Internet]. 2024 Jul. 13 [cited 2024 Aug. 31];87(2):194-201. Available from: https://revistas.upch.edu.pe/index.php/RNP/article/view/5254

Issue

Vol. 87 No. 2 (2024): Abril-junio

Section

CASE REPORTS

License

Copyright (c) 2024 Miguel A Vences, Maria Fe Albujar, Marcela Araujo, Diego Canales, Edu Cardenas, Ebelin Barja, Arturo Alvarez, Diego Urrunaga-Pastor

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This work is licensed under a Creative Commons Attribution-NonCommercial 4.0 International License.