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Open Access 06-09-2024 | Tocilizumab | Letter to the Editors

Highly beneficial outcome in severe acute necrotizing encephalopathy with tocilizumab treatment

Authors: Alexander Balck, Lara M. Lange, Alexander Neumann, Georg Royl, Philipp Jung, Jens Schaumberg, Norbert Brüggemann, Philipp J. Koch

Published in: Journal of Neurology

Excerpt

Acute necrotizing encephalopathy (ANE) is a severe neurologic condition that can arise following various systemic infections, including influenza and SARS-CoV-2 [1]. The outcome of ANE ranges from complete recovery (< 10%) to recovery of the acute episode with persistent deficits to death, with a mortality rate of up to 30% [2]. Affected individuals are, in most cases, young and present with rapid changes in consciousness, focal neurologic deficits, and epileptic seizures. Neuroimaging typically reveals symmetric, bilateral deep-gray matter lesions, often involving the thalami, with evidence of necrosis and/or hemorrhage [1]. …

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Shukla P, Mandalla A, Elrick MJ, Venkatesan A (2022) Clinical manifestations and pathogenesis of acute necrotizing encephalopathy: the interface between systemic infection and neurologic injury. Front Neurol 12:2. https://doi.org/10.3389/fneur.2021.628811CrossRef

Levine JM, Ahsan N, Ho E, Santoro JD (2020) Genetic acute necrotizing encephalopathy associated with RANBP2: clinical and therapeutic implications in pediatrics. Mult Scler Relat Disord 43:102194. https://doi.org/10.1016/j.msard.2020.102194CrossRefPubMedPubMedCentral

Ransohoff RM, Cardona AE (2010) The myeloid cells of the central nervous system parenchyma. Nature 468:253–262. https://doi.org/10.1038/nature09615CrossRefPubMed

Okumura A, Mizuguchi M, Kidokoro H et al (2009) Outcome of acute necrotizing encephalopathy in relation to treatment with corticosteroids and gammaglobulin. Brain Develop 31:221–227. https://doi.org/10.1016/j.braindev.2008.03.005CrossRef

Li G, Hilgenfeld R, Whitley R, De Clercq E (2023) Therapeutic strategies for COVID-19: progress and lessons learned. Nat Rev Drug Discov 22:449–475. https://doi.org/10.1038/s41573-023-00672-yCrossRefPubMedPubMedCentral

Huang Y, Zhou B, Hong S, Cai Y (2024) A case report and literature review on tocilizumab-cured acute necrotizing encephalopathy caused by influenza A virus. Front Pediatr. https://doi.org/10.3389/fped.2024.1351478CrossRefPubMedPubMedCentral

Koh JC, Murugasu A, Krishnappa J, Thomas T (2019) Favorable outcomes with early interleukin 6 receptor blockade in severe acute necrotizing encephalopathy of childhood. Pediatr Neurol 98:80–84. https://doi.org/10.1016/j.pediatrneurol.2019.04.009CrossRefPubMed

Yamamoto H, Okumura A, Natsume J et al (2015) A severity score for acute necrotizing encephalopathy. Brain Develop 37:322–327. https://doi.org/10.1016/j.braindev.2014.05.007CrossRef

Surtees R, DeSousa C (2006) Influenza virus associated encephalopathy. Arch Dis Child 91:455–456. https://doi.org/10.1136/adc.2005.092890CrossRefPubMedPubMedCentral

10.

Ito Y, Ichiyama T, Kimura H et al (1999) Detection of influenza virus RNA by reverse transcription-PCR and proinflammatory cytokines in influenza-virus-associated encephalopathy. J Med Virol 58:420–425. https://doi.org/10.1002/(SICI)1096-9071(199908)58:4%3c420::AID-JMV16%3e3.0.CO;2-TCrossRefPubMed

11.

Hoshino A, Takahashi N, Oka A, Mizuguchi M (2023) Association of IL6 and IL10 gene promotor polymorphisms with susceptibility to acute necrotizing encephalopathy. Front Neurosci. https://doi.org/10.3389/fnins.2023.1231957CrossRefPubMedPubMedCentral

12.

Mizuguchi M, Shibata A, Kasai M, Hoshino A (2023) Genetic and environmental risk factors of acute infection-triggered encephalopathy. Front Neurosci. https://doi.org/10.3389/fnins.2023.1119708CrossRefPubMedPubMedCentral

13.

Hosie PH, Lim C, Scott TRD et al (2023) Treatment of severe acute necrotizing encephalopathy of childhood with interleukin-6 receptor blockade in the first 24 h as add-on immunotherapy shows favorable long-term outcome at 2 years. Brain Dev 45:401–407. https://doi.org/10.1016/j.braindev.2023.03.002CrossRefPubMed

14.

Wang P-Y, Yang M-T, Liang J-S (2022) Acute necrotizing encephalopathy caused by SARS-CoV-2 in a child. Pediatr Neonatol 63:642–644. https://doi.org/10.1016/j.pedneo.2022.06.003CrossRefPubMedPubMedCentral

15.

Preuss CV, Anjum F. Tocilizumab. [Updated 2024 Feb 12]. In: StatPearls. Treasure Island (FL): StatPearls Publishing; 2024 Jan-. Available from: https://www.ncbi.nlm.nih.gov/books/NBK570644/

16.

Mizuguchi M, Ichiyama T, Imataka G et al (2021) Guidelines for the diagnosis and treatment of acute encephalopathy in childhood. Brain Develop 43:2–31. https://doi.org/10.1016/j.braindev.2020.08.001CrossRef

17.

Bloch C, Suter B, Fischmann A et al (2015) Only a touch of the flu? The simultaneous manifestation of acute necrotizing encephalopathy in two consanguineous patients. Open Forum Infect Dis 2:13. https://doi.org/10.1093/ofid/ofv013CrossRef

18.

Mizuguchi M (1997) Acute necrotizing encephalopathy of childhood: a novel form of acute encephalopathy prevalent in Japan and Taiwan. Brain Develop 19:81–92. https://doi.org/10.1016/S0387-7604(96)00063-0CrossRef

19.

Neilson DE, Adams MD, Orr CMD et al (2009) Infection-triggered familial or recurrent cases of acute necrotizing encephalopathy caused by mutations in a component of the nuclear pore, RANBP2. Am J Hum Genet 84:44–51. https://doi.org/10.1016/j.ajhg.2008.12.009CrossRefPubMedPubMedCentral

Title: Highly beneficial outcome in severe acute necrotizing encephalopathy with tocilizumab treatment
Authors: Alexander Balck
Lara M. Lange
Alexander Neumann
Georg Royl
Philipp Jung
Jens Schaumberg
Norbert Brüggemann
Philipp J. Koch
Publication date: 06-09-2024
Publisher: Springer Berlin Heidelberg
Keywords: Tocilizumab
Influenza
Encephalopathy
Published in: Journal of Neurology
Print ISSN: 0340-5354
Electronic ISSN: 1432-1459
DOI: https://doi.org/10.1007/s00415-024-12661-1

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