Publication Date

4-1-2025

Journal

Annals of Clinical and Translational Neurology

DOI

10.1002/acn3.70011

PMID

39932904

PMCID

PMC12040501

PubMedCentral® Posted Date

2-11-2025

PubMedCentral® Full Text Version

Post-print

Abstract

Febrile infection related epilepsy syndrome (FIRES) is a rare presentation of refractory status epilepticus with immune dysregulation as a potential pathologic mechanism. Despite promising results from second-line immunomodulators, approximately 30% remain refractory to treatment. We describe two children with FIRES who were unable to wean from anesthetic infusions with immunomodulatory treatment and subsequently received concurrent intrathecal dexamethasone and anakinra/tocilizumab as escalation of therapy. Following the initiation of this combined regimen, anesthetic infusions were decreased while maintaining seizure freedom. These cases demonstrate proof of principle that a multi-modal approach may be beneficial and should be considered in the treatment of FIRES.

Keywords

Child, Preschool, Humans, Male, Antibodies, Monoclonal, Humanized, Dexamethasone, Drug Resistant Epilepsy, Drug Therapy, Combination, Immunotherapy, Injections, Spinal, Interleukin 1 Receptor Antagonist Protein, Seizures, Febrile, Status Epilepticus, Child, anakinra, CNS immune modulation, febrile infection related epilepsy syndrome (FIRES), intrathecal dexamethasone, new onset refractory status epilepticus (NORSE), systemic immune modulation, tocilizumab

Published Open-Access

yes

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