Seizure control after late introduction of anakinra in a patient with adult onset Rasmussen's encephalitis.
ASM, antiseizure medication
Anakinra
Drug resistant epilepsy
FBTC, Focal to bilateral tonic-clonic
FIRES, febrile infection-related epilepsy syndrome
GTC, Generalized tonic-clonic
Hemiatrophy
IVIG, Intravenous immunoglobulins
Neuroinflammation
Rasmussen’s encephalitis
Journal
Epilepsy & behavior reports
ISSN: 2589-9864
Titre abrégé: Epilepsy Behav Rep
Pays: United States
ID NLM: 101750909
Informations de publication
Date de publication:
2021
2021
Historique:
received:
12
02
2021
revised:
05
05
2021
accepted:
08
05
2021
entrez:
30
6
2021
pubmed:
1
7
2021
medline:
1
7
2021
Statut:
epublish
Résumé
Neuroinflammation has been considered an important pathophysiological process involved in epileptogenesis and may provide possibilities for new treatment possibilities. We present the case of a 45-year-old female with drug resistant epilepsy and progressive right-sided cerebral hemiatrophy associated with adult onset Rasmussen's encephalitis. Over a period of 26 years, she was treated with 14 different antiseizure medications, intravenous immunoglobulins, glucocorticosteroids, underwent two operations with focal resection and subpial transections, and tried out trigeminal nerve stimulation. Extensive blood tests, including antibodies relevant for autoimmune encephalitis, and brain biopsy did not show any signs of neuroinflammation. Eventually, the patient received the interleukin-1 receptor antagonist, anakinra. Within 1-2 days after injection, seizure frequency decreased significantly, and, after one week, the seizures stopped completely. Anakinra treatment was continued for 2 months. Stopping medication led to a relapse of seizures after 2 weeks, with a frequency of up to 45 seizures per day. Reintroduction of anakinra led to rapid recovery. Treatment with anakinra was continued for 7 months. The treatment was discontinued in April 2020, and the patient has been completely seizure free since then. There have been no other changes in antiseizure medication.
Identifiants
pubmed: 34189453
doi: 10.1016/j.ebr.2021.100462
pii: S2589-9864(21)00036-8
pmc: PMC8219739
doi:
Types de publication
Case Reports
Langues
eng
Pagination
100462Informations de copyright
© 2021 The Author(s).
Déclaration de conflit d'intérêts
The authors declare that they have no known competing financial interests or personal relationships that could have appeared to influence the work reported in this paper.
Références
Epilepsy Curr. 2014 Jan;14(1 Suppl):3-7
pubmed: 24955068
Proc Natl Acad Sci U S A. 2000 Oct 10;97(21):11534-9
pubmed: 11016948
J Neuroimmunol. 2016 Sep 15;298:146-52
pubmed: 27609288
Neurology. 2003 Dec 23;61(12):1807-10
pubmed: 14694056
Brain Behav Immun. 2011 Oct;25(7):1281-9
pubmed: 21473909
Neurobiol Dis. 2013 Nov;59:183-93
pubmed: 23938763
Seizure. 2020 Oct;81:76-83
pubmed: 32769034
BMC Neurol. 2020 Sep 24;20(1):359
pubmed: 32972372
Autoimmun Rev. 2017 Aug;16(8):803-810
pubmed: 28572051
Nat Rev Drug Discov. 2012 Aug;11(8):633-52
pubmed: 22850787
Brain. 2005 Mar;128(Pt 3):454-71
pubmed: 15689357
Ann Neurol. 2016 Dec;80(6):939-945
pubmed: 27770579
Neurobiol Dis. 2009 Feb;33(2):171-81
pubmed: 19010416
Ann Neurol. 2005 Jan;57(1):152-5
pubmed: 15622539
Neurobiol Dis. 2017 Mar;99:12-23
pubmed: 27939857
J Neuroinflammation. 2018 Feb 09;15(1):38
pubmed: 29426321
Lancet Neurol. 2014 Feb;13(2):195-205
pubmed: 24457189
Neurotherapeutics. 2014 Apr;11(2):297-310
pubmed: 24639375
Nat Rev Neurol. 2019 Aug;15(8):459-472
pubmed: 31263255
Ann Neurol. 2012 Jul;72(1):82-90
pubmed: 22829270
Neurology. 2013 Jul 23;81(4):395-7
pubmed: 23794679
Nat Rev Neurol. 2009 Aug;5(8):458-62
pubmed: 19657347
J Neurosci. 1999 Jun 15;19(12):5054-65
pubmed: 10366638
J Neuroinflammation. 2015 Jul 02;12:129
pubmed: 26133170