ArticleBMC infectious diseases2022
COVID-19 related acute necrotizing encephalopathy with extremely high interleukin-6 and RANBP2 mutation in a patient with recently immunized inactivated virus vaccine and no pulmonary involvement.
Article in BMC infectious diseases, 2022. The graph could read no effect estimate from its abstract, so it casts no vote on the map. It is linked to trial NCT05582512 (Clinical Characteristics and Long Term Impact on Pediatric COVID-19 in Taiwan), which is not on this map. Cited by 12 papers.
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Clinical Characteristics and Long Term Impact on Pediatric COVID-19 in Taiwan
Who cites it
12 citing papers in PubMed, 20 citations in OpenAlex.
- Beyond correlation and causation: epistemic caution, anomalous findings, and COVID-19 vaccine safety.Philosophy, ethics, and humanities in medicine : PEHM · 2026Review
- A pilot study of peripheral blood tsRNA expression profiling in children with influenza-associated acute necrotizing encephalopathy.Virology journal · 2026Article
- Two mild phenotype molybdenum cofactor deficiency patients with novel MOCS2 mutation and immunological treatment after COVID-19 infection.BMC neurology · 2026Article
- Serum IL-17A and IL-6 in paediatricEmerging microbes & infections · 2024Article
- COVID-19: Unveiling the Neuropsychiatric Maze-From Acute to Long-Term Manifestations.Biomedicines · 2024Review
- Clinical characteristics and genetic analysis of children with Omicron BF.7.14 type novel coronavirus-related acute necrotizing encephalopathy.Frontiers in neurology · 2024Article
- A case report and literature review on tocilizumab-cured acute necrotizing encephalopathy caused by influenza A virus.Frontiers in pediatrics · 2024Article
- Recurrent infection triggered encephalopathy syndrome in a pediatric patient withPediatric investigation · 2023Article
- Biallelic variants in ribonuclease inhibitor (RNH1), an inflammasome modulator, are associated with a distinctive subtype of acute, necrotizing encephalopathy.Genetics in medicine : official journal of the American College of Medical Genetics · 2023Article
- Neurological Adverse Reactions to SARS-CoV-2 Vaccines.Clinical psychopharmacology and neuroscience : the official scientific journal of the Korean College of Neuropsychopharmacology · 2023Review
- The youngest infant with COVID-19-associated necrotizing encephalitis in Asia: A case report.SAGE open medical case reports · 2023Article
- Clinical features, prognostic factors of pediatric acute necrotizing encephalopathy: a 67-case retrospective analysis.Frontiers in neurologyArticle
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Authors and funding
6 authors at 3 institutions in 2 countries.
Funding
No grant is acknowledged in the PubMed record.
Abstract
backgroundWe report the first case of COVID-19 associated acute necrotizing encephalopathy (ANE) without pulmonary disease in a patient with an extremely high interleukin-6 (IL-6) level and Ran Binding Protein 2 (RANBP2) mutation. CASE PRESENTATION: A 29-year-old woman recently immunized with inactivated viral vaccine-BBIBP32-CorV (Sinopharm) presented with alteration of consciousness. Her body temperature was 37° Celsius, blood pressure 42/31 mmHg, heart rate 130 bpm, respiratory rate 20 per minute, and oxygen saturation 98%. Respiratory examination was unremarkable. Neurological examination revealed stupor but preserved brainstem reflexes. Non-contrast computerized tomography of the brain showed symmetrical hypodense lesions involving bilateral thalami and cerebellar hemispheres characteristic of ANE. No pulmonary infiltration was found on chest radiograph. SARS-CoV-2 was detected by PCR; whole genome sequencing later confirmed the Delta variant. RANBP2 gene analysis revealed heterozygous Thr585Met mutation. Serum IL-6 was 7390 pg/mL. Urine examination showed pyelonephritis. Her clinical course was complicated by seizure, septic shock, acute kidney injury, and acute hepatic failure. She later developed coma and passed away in 6 days.
conclusionsANE is caused by cytokine storm leading to necrosis and hemorrhage of the brain. IL-6 was deemed as a prognostic factor and a potential treatment target of ANE in previous studies. RANBP2 missense mutation strongly predisposes this condition by affecting mitochondrial function, viral entry, cytokine signaling, immune response, and blood-brain barrier maintenance. Also, inactivated vaccine has been reported to precipitate massive production of cytokines by antibody dependent enhancement (ADE). The true incidence of COVID-19 associated ANE is not known as were the predictors of its development. We proposed these potential two factors (RANBP2 mutation and ADE) that could participate in the pathogenesis of ANE in COVID-19 apart from SARS-CoV2 infection by itself. Further study is needed to confirm this hypothesis, specifically in the post-vaccination period. Role of RANBP2 mutation and its application in COVID-19 and ANE should be further elaborated.
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