Acute haemorrhagic necrotizing encephalopathy in the setting of SARS-CoV-2 vaccination: a case report and literature review

Siriratnam, Pakeeran; Buzzard, Katherine; Yip, Gary

doi:10.1007/s13760-021-01862-4

Cited by 8 publications

(9 citation statements)

References 6 publications

Supporting

Mentioning

Contrasting

Order By: Relevance

“…The only potential precipitating factor was the administration of the ChAdOx1 nCoV-19 vaccine 2 days prior. Interestingly, there is a recently published case of a 75-year-old woman who developed ANE 2 days after receiving the ChAdOx1 nCoV-19 vaccine (15). Unlike our patient, however, the patient's outcome was unfavorable, as she died 1 month after disease onset despite corticosteroid treatment.…”

Section: Unfulfilledcontrasting

confidence: 59%

“…A case of ANE 6 days after diphtheria, tetanus toxoid, and wholecell pertussis (DTPw) vaccination was reported in a previously healthy 6-month-old boy (21). Since our case shares similar timing to the previously reported case of ANE (15), and the fact that ANE is not considered a demyelinating condition, we consider the timing of the vaccine to be plausible. Furthermore, the ChAdOx1 nCoV-19 vaccine uses an adenovirus vector (22).…”

Section: Unfulfilledmentioning

confidence: 52%

See 1 more Smart Citation

Case Report: Acute Necrotizing Encephalopathy Following COVID-19 Vaccine

et al. 2022

View full text Add to dashboard Cite

ObjectivesAcute necrotizing encephalopathy (ANE) is a rare neurological disorder arising from a para- or post-infectious “cytokine storm. ”It has recently been reported in association with coronavirus disease 2019 (COVID-19) infection.MethodsA 56-year-old male with a diagnosis of ANE 48 h following the first dose of ChAdOx1 nCoV-19 vaccination was investigated. Cytokine analyses on serum and cerebrospinal fluid (CSF) were performed. The patient was treated with high-dose corticosteroids and followed clinically and radiologically.ResultsFavorable clinical and radiological outcomes were noted. There was an upregulation in serum levels of CXCL5, CXCL1, Il-8, IL-15, CCL2, TGF-B, and EGF, and up-regulation in CSF levels of CXCL5, IL-2, IL-3, and IL-8.DiscussionAs COVID-19 infection has been previously reported as a possible rare cause of ANE, we speculate on an aberrant immune response mechanism that was brought about by the vaccine. To increase our understanding of the pathogenesis of ANE in the context of COVID-19 vaccination and to better define its clinical features and outcomes, clinicians and scientists should continue reporting convincing cases of such entities.

show abstract

Section: Unfulfilledcontrasting

confidence: 59%

Section: Unfulfilledmentioning

confidence: 52%

Case Report: Acute Necrotizing Encephalopathy Following COVID-19 Vaccine

et al. 2022

View full text Add to dashboard Cite

show abstract

“…She received her first dose of the ChAdOx1nCoV-1 vaccine 2 days prior to hospitalization and was also on a course of cephalexin for a presumed UTI. Testing of the patient’s serum for SARS-CoV-2 antibodies was negative [ 7 ]. Apart from cytokine storm, gene mutation has also been reported to precipitate ANE [ 2 ].…”

Section: Discussionmentioning

confidence: 99%

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

et al. 2022

View full text Add to dashboard Cite

Background We 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. Conclusions ANE 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.

show abstract

“…These included acute encephalopathy ( Roberts et al, 2021 , Grover et al, 2022 , Al-Mashdali et al, 2021 , Baldelli et al, 2021 ) or encephalitis, ( Zuhorn et al, 2021 , Torrealba-Acosta et al, 2021 , Fan et al, 2022 , Kobayashi et al, 2022 , Zlotnik et al, 2021 , Takata et al, 2021 ) which in some cases were associated with seizures including two cases of new-onset refractory status epilepticus (NORSE), ( Fan et al, 2022 , Aladdin and Shirah, 2021 ). There are also several reports of acute disseminated encephalomyelitis (ADEM) or ADEM-like presentations ( Vogrig et al, 2021 , Cao and Ren, 2021, Ozgen Kenangil et al, 2021 , Rinaldi et al, 2021, Kania et al, 2021 ) and acute necrotizing encephalopathy ( Bensaidane et al, 2022 , Siriratnam et al, 2022 ). In almost all of the aforementioned cases, when EEG findings were reported, they consisted of nonspecific generalized slow activity ( Zuhorn et al, 2021 , Torrealba-Acosta et al, 2021 , Fan et al, 2022 , Aladdin and Shirah, 2021 , Al-Mashdali et al, 2021 , Baldelli et al, 2021 , Vogrig et al, 2021 , Bensaidane et al, 2022 , Siriratnam et al, 2022 ) or were assessed as normal ( Sluyts et al, 2022 , Zlotnik et al, 2021 , Takata et al, 2021 , Grover et al, 2022 ).…”

Section: Electroencephalography (Eeg)mentioning

confidence: 99%

“…There are also several reports of acute disseminated encephalomyelitis (ADEM) or ADEM-like presentations ( Vogrig et al, 2021 , Cao and Ren, 2021, Ozgen Kenangil et al, 2021 , Rinaldi et al, 2021, Kania et al, 2021 ) and acute necrotizing encephalopathy ( Bensaidane et al, 2022 , Siriratnam et al, 2022 ). In almost all of the aforementioned cases, when EEG findings were reported, they consisted of nonspecific generalized slow activity ( Zuhorn et al, 2021 , Torrealba-Acosta et al, 2021 , Fan et al, 2022 , Aladdin and Shirah, 2021 , Al-Mashdali et al, 2021 , Baldelli et al, 2021 , Vogrig et al, 2021 , Bensaidane et al, 2022 , Siriratnam et al, 2022 ) or were assessed as normal ( Sluyts et al, 2022 , Zlotnik et al, 2021 , Takata et al, 2021 , Grover et al, 2022 ). Thus, these data indicate that there is no specific EEG pattern that affirms an association of vaccination against SARS-Covid-2 with post-vaccination neurological dysfunction.…”

Section: Electroencephalography (Eeg)mentioning

confidence: 99%