Document Type : Case Report
Authors
1 Department of Pediatric Neurology, Ali Asghar Children’s Hospital, Iran University of Medical Sciences, Tehran, Iran
2 Department of Pediatric Psychiatry, Ali Asghar Children's Hospital, Iran University of Medical Sciences, Tehran, Iran
3 Pediatrician, Iran University of Medical Sciences, Tehran, Iran
Abstract
Background: Autoimmune encephalitis (AIE) with neural cell autoantibodies is a treatable category of non-viral encephalitis in children and adolescents which presents with neuropsychiatric manifestations. Anti-N-methyl- D-aspartate receptor encephalitis, the most common identified type of AIE, is more frequent than individual viral encephalitis in young patients. Early diagnosis and treatment of the disease have been associated with improvement in the majority of patients.
Case Report: we report two pediatric cases with acute neuropsychiatric presentations in whom the diagnosis of AIE was proved based on the cerebrospinal fluid (CSF) antineuronal antibodies in one and on clinical and paraclinical grounds in another seronegative patient. Immunotherapy in both patients resulted in improvement. No underlying tumor was found in patients.
Pediatricians should be aware of manifestations, investigations, and treatment of AIE and consider it as the differential diagnosis of encephalitis.
Keywords
- Armangue T, Petit-Pedrol M, Dalmau J. Autoimmune encephalitis in children. J Child Neurol 2012; 27(11):1460-9. doi: 10.1177/0883073812448838.
- Brenton JN, Goodkin HP. Antibody-mediated autoimmune encephalitis in childhood. Pediatr Neurol 2016; 60:13-23. doi: 10.1016/j.pediatrneurol.2016.04.004.
- Dalmau J, Graus F. Antibody-mediated encephalitis. N Engl J Med 2018; 378(9):840-51. doi: 10.1056/NEJMra1708712.
- Dutra LA, Abrantes F, Toso FF, Pedroso JL, Barsottini OG, Hoftberger R. Autoimmune encephalitis: a review of diagnosis and treatment. Arq Neuropsiquiatr 2018; 76(1):41-9. doi: 10.1590/0004-282X20170176.
- Barbagallo M, Vitaliti G, Pavone P, Romano C, Lubrano R, Falsaperla R. Pediatric autoimmune encephalitis. J Pediatr Neurosci 2017; 12(2):130-4. doi: 10.4103/jpn.JPN_185_16.
- 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. doi: 10.1016/S1474-4422(12)70310-1.
- Stingl C, Cardinale K, Van Mater H. An update on the treatment of pediatric autoimmune encephalitis. Curr Treatm Opt Rheumatol 2018; 4(1):14-28. doi: 10.1007/s40674-018-0089-z.
- Florance NR, Davis RL, Lam C, Szperka C, Zhou L, Ahmad S, et al. Anti-N-methyl-D-aspartate receptor (NMDAR) encephalitis in children and adolescents. Ann Neurol 2009; 66(1):11-8. doi: 10.1002/ana.21756.
- Moscato EH, Peng X, Jain A, Parsons TD, Dalmau J, Balice-Gordon RJ. Acute mechanisms underlying antibody effects in anti-N-methyl-D-aspartate receptor encephalitis. Ann Neurol 2014; 76(1):108-19. doi: 10.1002/ana.24195.
- Luca N, Daengsuwan T, Dalmau J, Jones K, deVeber G, Kobayashi J, et al. Anti-N-methyl-D-aspartate receptor encephalitis: a newly recognized inflammatory brain disease in children. Arthritis Rheum 2011; 63(8):2516-22. doi: 10.1002/art.30437.
- Kelley BP, Patel SC, Marin HL, Corrigan JJ, Mitsias PD, Griffith B. Autoimmune encephalitis: pathophysiology and imaging review of an overlooked diagnosis. AJNR Am J Neuroradiol 2017; 38(6):1070-8. doi: 10.3174/ajnr.A5086.
- Probasco JC, Solnes L, Nalluri A, Cohen J, Jones KM, Zan E, et al. Abnormal brain metabolism on FDG-PET/CT is a common early finding in autoimmune encephalitis. Neurol Neuroimmunol Neuroinflamm 2017; 4(4):e352. doi: 10.1212/NXI.0000000000000352.
- Dale RC, Gorman MP, Lim M. Autoimmune encephalitis in children: clinical phenomenology, therapeutics, and emerging challenges. Curr Opin Neurol 2017; 30(3):334-44. doi: 10.1097/WCO.0000000000000443.
- Paterson RW, Zandi MS, Armstrong R, Vincent A, Schott JM. Clinical relevance of positive voltage-gated potassium channel (VGKC)-complex antibodies: experience from a tertiary referral centre. J Neurol Neurosurg Psychiatry 2014; 85(6):625-30. doi: 10.1136/jnnp-2013-305218.
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