Article ; Online: Factors associated with outcomes of severe acute necrotizing encephalopathy: A multicentre experience in Malaysia.
Developmental medicine and child neurology
2023 Volume 65, Issue 9, Page(s) 1256–1263
Abstract: This case series compared clinical variables and various combinations of immunotherapy received with outcomes of patients with severe acute necrotizing encephalopathy (ANE). We performed a retrospective review of clinical variables, immunotherapy ... ...
Abstract | This case series compared clinical variables and various combinations of immunotherapy received with outcomes of patients with severe acute necrotizing encephalopathy (ANE). We performed a retrospective review of clinical variables, immunotherapy received, and outcomes (based on the modified Rankin Scale) in Malaysia between February 2019 and January 2020. Twenty-seven children (12 male), aged 7 months to 14 years (mean 4 years) at diagnosis were included. Of these, 23 had an ANE severity score of 5 to 9 out of 9 (high risk). Eleven patients received tocilizumab (four in combination with methylprednisolone [MTP], seven with MTP + intravenous immunoglobulin [IVIG]) and 16 did not (two received MTP alone, 14 received MTP + IVIG). Nine died. Among the survivors, six had good outcomes (modified Rankin Score 0-2) at 6 months follow-up. All patients who received tocilizumab in combination with MTP + IVIG survived. Twenty children received first immunotherapy within 48 hours of admission. No significant association was found between the timing of first immunotherapy with outcomes. Those with brainstem dysfunction (p = 0.016) were observed to have poorer outcomes. This study showed a trend towards better survival when those with severe ANE were treated with tocilizumab in combination with MTP + IVIG. However, larger studies will be needed to determine the effect of this regime on the long-term outcomes. |
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MeSH term(s) | Child ; Humans ; Male ; Immunoglobulins, Intravenous/therapeutic use ; Malaysia ; Methylprednisolone ; Leukoencephalitis, Acute Hemorrhagic/therapy ; Brain Diseases ; Retrospective Studies |
Chemical Substances | Immunoglobulins, Intravenous ; Methylprednisolone (X4W7ZR7023) |
Language | English |
Publishing date | 2023-02-07 |
Publishing country | England |
Document type | Multicenter Study ; Case Reports |
ZDB-ID | 80369-8 |
ISSN | 1469-8749 ; 0012-1622 |
ISSN (online) | 1469-8749 |
ISSN | 0012-1622 |
DOI | 10.1111/dmcn.15536 |
Database | MEDical Literature Analysis and Retrieval System OnLINE |
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