Seizure control after late introduction of anakinra in a patient with adult onset Rasmussen’s encephalitis

Mochol, Monika; Taubøll, Erik; Sveberg, Line; Tennøe, Bjørn; Olsen, Ketil Berg; Svalheim, Sigrid

doi:10.1016/j.ebr.2021.100462

Cited by 14 publications

(7 citation statements)

References 25 publications

(36 reference statements)

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“…Anakinra, an anti-IL1 receptor, has shown antiepileptic properties in patients with super refractory epileptic status [110], RE [111], and especially in cases of febrile infection-related epilepsy syndrome (FIRES), for which it has been proposed as a firstline therapeutic alternative [112,113]. Additionally, anakinra has been shown to reduce seizures in animal models of anti-NMDAR encephalitis and lithium-pilocarpineinduced epilepsy.…”

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confidence: 99%

Immune Mechanism of Epileptogenesis and Related Therapeutic Strategies

et al. 2022

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Immunologic and neuroinflammatory pathways have been found to play a major role in the pathogenesis of many neurological disorders such as epilepsy, proposing the use of novel therapeutic strategies. In the era of personalized medicine and in the face of the exhaustion of anti-seizure therapeutic resources, it is worth looking at the current or future possibilities that neuroimmunomodulator or anti-inflammatory therapy can offer us in the management of patients with epilepsy. For this reason, we performed a narrative review on the recent advances on the basic epileptogenic mechanisms related to the activation of immunity or neuroinflammation with special attention to current and future opportunities for novel treatments in epilepsy. Neuroinflammation can be considered a universal phenomenon and occurs in structural, infectious, post-traumatic, autoimmune, or even genetically based epilepsies. The emerging research developed in recent years has allowed us to identify the main molecular pathways involved in these processes. These molecular pathways could constitute future therapeutic targets for epilepsy. Different drugs current or in development have demonstrated their capacity to inhibit or modulate molecular pathways involved in the immunologic or neuroinflammatory mechanisms described in epilepsy. Some of them should be tested in the future as possible antiepileptic drugs.

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confidence: 99%

Immune Mechanism of Epileptogenesis and Related Therapeutic Strategies

et al. 2022

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“…One paper on multifocal neutrophilic meningoencephalitis (MNM) was excluded as did not directly analyze the effect on the epileptic phenotype (104). Twelve articles were finally included in this review (Figure 3), for a total of 37 cases of epilepsy with different etiologies (105)(106)(107)(108)(109)(110)(111)(112)(113)(114)(115)(116). All publications were in English, six were from the USA, three were from Europe, one from Asia, and two were international studies, involving different countries.…”

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confidence: 99%

Targeting Inflammatory Mediators in Epilepsy: A Systematic Review of Its Molecular Basis and Clinical Applications

et al. 2022

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IntroductionRecent studies prompted the identification of neuroinflammation as a potential target for the treatment of epilepsy, particularly drug-resistant epilepsy, and refractory status epilepticus. This work provides a systematic review of the clinical experience with anti-cytokine agents and agents targeting lymphocytes and aims to evaluate their efficacy and safety for the treatment of refractory epilepsy. Moreover, the review analyzes the main therapeutic perspectives in this field.MethodsA systematic review of the literature was conducted on MEDLINE database. Search terminology was constructed using the name of the specific drug (anakinra, canakinumab, tocilizumab, adalimumab, rituximab, and natalizumab) and the terms “status epilepticus,” “epilepsy,” and “seizure.” The review included clinical trials, prospective studies, case series, and reports published in English between January 2016 and August 2021. The number of patients and their age, study design, specific drugs used, dosage, route, and timing of administration, and patients outcomes were extracted. The data were synthesized through quantitative and qualitative analysis.ResultsOur search identified 12 articles on anakinra and canakinumab, for a total of 37 patients with epilepsy (86% febrile infection-related epilepsy syndrome), with reduced seizure frequency or seizure arrest in more than 50% of the patients. The search identified nine articles on the use of tocilizumab (16 patients, 75% refractory status epilepticus), with a high response rate. Only one reference on the use of adalimumab in 11 patients with Rasmussen encephalitis showed complete response in 45% of the cases. Eight articles on rituximab employment sowed a reduced seizure burden in 16/26 patients. Finally, one trial concerning natalizumab evidenced a response in 10/32 participants.ConclusionThe experience with anti-cytokine agents and drugs targeting lymphocytes in epilepsy derives mostly from case reports or series. The use of anti-IL-1, anti-IL-6, and anti-CD20 agents in patients with drug-resistant epilepsy and refractory status epilepticus has shown promising results and a good safety profile. The experience with TNF inhibitors is limited to Rasmussen encephalitis. The use of anti-α4-integrin agents did not show significant effects in refractory focal seizures. Concerning research perspectives, there is increasing interest in the potential use of anti-chemokine and anti-HMGB-1 agents.

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“…Anakinra have also shown some positive effect in cases of NORSE/FIRES [79]. A case report on the use of anakinra in a patient with RE, 28 years after clinical onset, showed a complete cessation of seizures for a period of 13 months without recurrence [80] (table 6).…”

Section: Treatments Targeting Pro-inflammatory Cytokinesmentioning

confidence: 99%