Auto-immune anti-N-methyl-D-aspartate receptor encephalitis: A two-case report

Authors

  • Jorge André Soares Abreu Ferreira Serviço de Pediatria, Centro Hospitalar de Trás-os-Montes e Alto Douro, Vila Real, Portugal
  • Sara Cecilia Carneiro Peixoto Serviço de Pediatria, Centro Hospitalar de Trás-os-Montes e Alto Douro, Vila Real, Portugal
  • Inês Conceição Cancelo Carrilho Unidade de Neuropediatria, Centro Materno Infantil do Norte, Centro Hospitalar e Universitário do Porto
  • Vânia Sofia Vicente Martins Serviço de Pediatria, Centro Hospitalar de Trás-os-Montes e Alto Douro, Vila Real, Portugal

DOI:

https://doi.org/10.15448/1980-6108.2018.2.29534

Keywords:

encephalitis, anti-N-methyl-D-aspartate receptor encephalitis, anti-NMDA receptor encephalitis, seizures, movement disorders.

Abstract

AIMS: To report two cases with different presentations of anti-N-methyl-D-aspartate receptor (anti-NMDAR) encephalitis, a newly identified autoimmune disease characterized by consciousness changes, memory deficit, seizures, autonomic dysfunction and movement disorders.

CASES DESCRIPTION: A six-year-old female, who presented with dystonic and choreoathetoid movements with refusal to walk. A 17-year-old male, presented with behavioral changes, retrograde amnesia and seizures. Electroencephalogram and brain magnetic resonance imaging did not show any significant findings. Anti-NMDAR antibodies were positive in cerebrospinal fluid in both cases and in serum in the first patient. Methylprednisolone, immunoglobulin and rituximab were given at different intervals. Both had a recurrence about six months later, with recovery at the end of one and a half year of the diagnosis.

CONCLUSIONS: Anti-NMDAR encephalitis should be considered in patients with sudden onset of neuropsychiatric symptoms. Early diagnosis and treatment are major prognostic factors.

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References

Goenka A, Jain V, Nariai H, Spiro A, Steinschneider M. Extended Clinical Spectrum of Anti-N-Methyl-d-Aspartate Receptor Encephalitis in Children: A Case Series. Pediatr Neurol. 2017;72:51-5. https://doi.org/10.1016/j.pediatrneurol.2017.03.010

Borlot F, Santos ML, Bandeira M, Liberalesso PB, Kok F, Löhr A Jr, Reed UM. Anti-N-methyl D-aspartate receptor encephalitis in childhood. J Pediatr (Rio J). 2012;88(3):275-8. https://doi.org/10.2223/JPED.2172

Kahn I, Helman G, Vanderver A, Wells E. Anti- N-Methyl-d-Aspartate (NMDA) Receptor Encephalitis. J Child Neurol. 2017;32(2):243-5. https://doi.org/10.1177/0883073816675557

Dalmau J, Tüzün E, Wu HY, Masjuan J, Rossi JE, Voloschin A, Baehring JM, Shimazaki H, Koide R, King D, Mason W, Sansing LH, Dichter MA, Rosenfeld MR, Lynch DR. Paraneoplastic anti-N-methyl-D-aspartate receptor encephalitis associated with ovarian teratoma. Ann Neurol. 2007;61:25-36. https://doi.org/10.1002/ana.21050

Liu CY, Zhu J, Zheng XY, Ma C, Wang X. Anti-N-Methyl-D-aspartate Receptor Encephalitis: A Severe, Potentially Reversible Autoimmune Encephalitis. Mediators Inflamm. 2017; 2017:6361479. https://doi.org/10.1155/2017/6361479

Gable MS, Sheriff H, Dalmau J, Tilley DH, Glaser CA. The frequency of autoimmune N-methyl-D-aspartate receptor encephalitis surpasses that of individual viral etiologies in young individuals enrolled in the California Encephalitis Project. Clin Infect Dis. 2012;54:899-904. https://doi.org/10.1093/cid/cir1038

Matoq AA1, Rappoport AS, Yang Y, O'Babatunde J, Bakerywala R, Sheth RD. Anti-NMDA-receptor antibody encephalitis in infants. Epilepsy Behav Case Rep. 2015 Nov 4;4:99-101. https://doi.org/10.1016/j.ebcr.2015.07.005

Goldberg EM, Titulaer M, de Blank PM, Sievert A, Ryan N. Anti-N-methyl-D-aspartate receptor-mediated encephalitis in infants and toddlers: case report and review of the literature. Pediatr Neurol. 2014;50(2):181-4. https://doi.org/10.1016/j.pediatrneurol.2013.10.002

Bashiri FA, Al-Rasheed AA, Hassan SM, Hamad MHA, El Khashab HY, Kentab AY, AlBadr FB, Salih MA. Auto-immune anti-N-methyl-D-aspartate receptor (anti-NMDAR) encephalitis: three case reports, Paediatr Int Child Health. 2017;37(3):222-6. https://doi.org/10.1080/20469047.2016.1191852

Titulaer MJ, McCracken L, Gabilondo I, Armangué T, Glaser C, Iizuka T, Honig LS, Benseler SM, Kawachi I, Martinez-Hernandez E, Aguilar E, Gresa-Arribas N, Ryan-Florance N, Torrents A, Saiz A, Rosenfeld MR, Balice-Gordon R, Graus F, Dalmau J. Treatment and prognostic factors for long-term outcome in patients with anti-N-Methyl-D-Aspartate (NMDA) receptor encephalitis: a cohort study. Lancet neurology. 2013;12(2):157-65. https://doi.org/10.1016/S1474-4422(12)70310-1

Brenton JN, Kim J, Schwartz RH. Approach to the Management of Pediatric-Onset Anti-N-Methyl-d-Aspartate (Anti-NMDA) Receptor Encephalitis: A Case Series. J Child Neurol. 2016;31(9):1150-5. https://doi.org/10.1177/0883073816643406

Published

2018-02-27

How to Cite

Ferreira, J. A. S. A., Peixoto, S. C. C., Carrilho, I. C. C., & Martins, V. S. V. (2018). Auto-immune anti-N-methyl-D-aspartate receptor encephalitis: A two-case report. Scientia Medica, 28(2), ID29534. https://doi.org/10.15448/1980-6108.2018.2.29534

Issue

Section

Case Reports