Thalamic Lesions in a Toddler with Glutamic Acid Decarboxylase Autoimmune Encephalitis

Abstract

We present a child with glutamic acid decarboxylase (GAD) autoimmune encephalitis (AE) with bilateral thalamic lesions on magnetic resonance imaging (MRI) of the brain. A healthy 21-month-old girl, after superrefractory status epilepticus (SE) and fever developed dyskinesia and ataxia, which we subsequently diagnosed as GAD AE. She showed remarkable response to treatment with methylprednisolone and intravenous immunoglobulin (IVIg). Following an initial normal MRI of her brain, a further brain imaging showed bilateral thalamic lesions. This is an unusual finding since brain imaging abnormalities, when found in GAD AE, mostly involve the mesial temporal lobe structures. Thalamic lesions in GAD AE have not been reported previously.

Keywords

GAD - limbic encephalitis - bilateral thalamic lesions - status epilepticus

Publikationsverlauf

Eingereicht: 21. Juni 2020

Angenommen: 18. August 2020

Artikel online veröffentlicht:
21. September 2020

• References

• 1 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 (06) 1070-1078
• 2 Bien CG, Bien CI. Autoimmune encephalitis in children and adolescents. Neurol Res Pract 2020;2(04). Doi: https://doi.org/10.1186/s42466-019-0047-8
• 3 Cellucci T, Van Mater H, Graus F. et al. Clinical approach to the diagnosis of autoimmune encephalitis in the pediatric patient. Neurol Neuroimmunol Neuroinflamm 2020; 7 (02) e663
• 4 Graus F, Titulaer MJ, Balu R. et al. A clinical approach to diagnosis of autoimmune encephalitis. Lancet Neurol 2016; 15 (04) 391-404
• 5 Ward RJ, Varughese GI, Jose B, Abraham RJ. Lesson of the month 2: Autoimmune sequelae of anti-GAD antibodies - thinking outside the box. Clin Med (Lond) 2017; 17 (05) 473-474
• 6 Solimena M, De Camilli P. Autoimmunity to glutamic acid decarboxylase (GAD) in Stiff-Man syndrome and insulin-dependent diabetes mellitus. Trends Neurosci 1991; 14 (10) 452-457
• 7 Giometto B, Nicolao P, Macucci M, Tavolato B, Foxon R, Bottazzo GF. Temporal-lobe epilepsy associated with glutamic-acid-decarboxylase autoantibodies. Lancet 1998; 352: 457
• 8 Saiz A, Blanco Y, Sabater L. et al. Spectrum of neurological syndromes associated with glutamic acid decarboxylase antibodies: diagnostic clues for this association. Brain 2008; 131 (Pt 10): 2553-2563
• 9 Ariño H, Gresa-Arribas N, Blanco Y. et al. Cerebellar ataxia and glutamic acid decarboxylase antibodies: immunologic profile and long-term effect of immunotherapy. JAMA Neurol 2014; 71 (08) 1009-1016
• 10 Triplett J, Vijayan S, MacDonald A. et al. Fulminant anti-GAD antibody encephalitis presenting with status epilepticus requiring aggressive immunosuppression. J Neuroimmunol 2018; 323: 119-124
• 11 Korff CM, Parvex P, Cimasoni L. et al. Encephalitis associated with glutamic acid decarboxylase autoantibodies in a child: a treatable condition?. Arch Neurol 2011; 68 (08) 1065-1068
• 12 Kairamkonda S, Kapil K, Gabriel G, Veerapandiyan A. A challenging case of intractable seizures in a pediatric patient with anti-GAD65 encephalitis. Neurology 2019; 92 (15 Supplement): P2.2-019
• 13 Brenton JN, Goodkin HP. Antibody-mediated autoimmune encephalitis in childhood. Pediatr Neurol 2016; 60: 13-23
• 14 Malter MP, Helmstaedter C, Urbach H, Vincent A, Bien CG. Antibodies to glutamic acid decarboxylase define a form of limbic encephalitis. Ann Neurol 2010; 67 (04) 470-478
• 15 Spatola M, Novy J, Du Pasquier R, Dalmau J, Rossetti AO. Status epilepticus of inflammatory etiology: a cohort study. Neurology 2015; 85 (05) 464-470
• 16 de Bruijn MAAM, Bruijstens AL, Bastiaansen AEM. et al; CHANCE Study Group. Pediatric autoimmune encephalitis: recognition and diagnosis. Neurol Neuroimmunol Neuroinflamm 2020; 7 (03) e682
• 17 Akman CI, Patterson MC, Rubinstein A, Herzog R. Limbic encephalitis associated with anti-GAD antibody and common variable immune deficiency. Dev Med Child Neurol 2009; 51 (07) 563-567
• 18 Mishra N, Rodan LH, Nita DA, Gresa-Arribas N, Kobayashi J, Benseler SM. Anti-glutamic acid decarboxylase antibody associated limbic encephalitis in a child: expanding the spectrum of pediatric inflammatory brain diseases. J Child Neurol 2014; 29 (05) 677-683
• 19 Blanc F, Ruppert E, Kleitz C. et al. Acute limbic encephalitis and glutamic acid decarboxylase antibodies: a reality?. J Neurol Sci 2009; 287 (1-2): 69-71
• 20 Renard D, Castelnovo G, Campello C. et al. Thalamic lesions: a radiological review. Behav Neurol 2014; 2014: 154631
• 21 Lyon JB, Remigio C, Milligan T, Deline C. Acute necrotizing encephalopathy in a child with H1N1 influenza infection. Pediatr Radiol 2010; 40 (02) 200-205
• 22 Gualtieri F, Curia G, Marinelli C, Biagini G. Increased perivascular laminin predicts damage to astrocytes in CA3 and piriform cortex following chemoconvulsive treatments. Neuroscience 2012; 218: 278-294
• 23 Costa AM, Lucchi C, Simonini C, Rosal Lustosa Í, Biagini G. Status epilepticus dynamics predicts latency to spontaneous seizures in the kainic acid model. Cell Physiol Biochem 2020; 54 (03) 493-507