Developmental medicine and child neurology · 2026
Early tocilizumab and outcomes in acute necrotizing encephalopathy.
СтатьяНазвание и аннотация: Перевод в очереди
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Han Velda X, Poudyal Prithuja, Lee Vanessa, Saini Arushi Gahlot, Mohammad Shekeeb S, Pillai Sekhar C, Passi Gouri Rao, Sankhyan Naveen, Mohamed Ahmad Rithauddin, Lingappa Lokesh, Sakuma Hiroshi, Dale Russell C, Lim Ming, Thomas Terrence, International ANE Study Group
Аннотация
AIM: To evaluate the timing and safety of tocilizumab in acute necrotizing encephalopathy (ANE).
METHOD: This was a multicentre retrospective study evaluating outcomes (modified Rankin Scale [mRS]) in children with ANE treated with tocilizumab. A logistic regression model determined a cut-off for outcomes (binary good [mRS 0-2] vs poor [mRS 3-5]) according to day of tocilizumab administration, enabling a dichotomy of 'early' and 'late' treatment groups. Expected outcomes according to ANE severity score (ANE-SS) in early-treatment and late-treatment groups were compared against reference data (conventional therapy, no tocilizumab) for outcomes according to risk category (medium risk: ANE-SS score 2-4; high risk: ANE-SS score ≥5).
RESULTS: From 2018 to 2023, 63 children (mean age 6 years 0 months [range: 8 months-15 years 4 months]; 33 males, 30 females) were treated with tocilizumab, alongside corticosteroids in all and intravenous immunoglobulin in 33 (52.4%). Logistic regression defined an optimal treatment window of ≤3.3 days. Compared to the reference data, early tocilizumab (≤ 72 hours after onset, n = 49) was associated with good outcomes in both high-risk (OR = 11.43, 95% confidence interval [CI] = 1.64-127.4, p = 0.01) and medium-risk (OR = 12.80, 95% CI = 2.15-67.8; p = 0.006) ANE-SS categories. No tocilizumab-related adverse events were reported.
INTERPRETATION: Early tocilizumab treatment was associated with improved outcomes in severe ANE, with no adverse events, compared to conventional therapy alone.
METHOD: This was a multicentre retrospective study evaluating outcomes (modified Rankin Scale [mRS]) in children with ANE treated with tocilizumab. A logistic regression model determined a cut-off for outcomes (binary good [mRS 0-2] vs poor [mRS 3-5]) according to day of tocilizumab administration, enabling a dichotomy of 'early' and 'late' treatment groups. Expected outcomes according to ANE severity score (ANE-SS) in early-treatment and late-treatment groups were compared against reference data (conventional therapy, no tocilizumab) for outcomes according to risk category (medium risk: ANE-SS score 2-4; high risk: ANE-SS score ≥5).
RESULTS: From 2018 to 2023, 63 children (mean age 6 years 0 months [range: 8 months-15 years 4 months]; 33 males, 30 females) were treated with tocilizumab, alongside corticosteroids in all and intravenous immunoglobulin in 33 (52.4%). Logistic regression defined an optimal treatment window of ≤3.3 days. Compared to the reference data, early tocilizumab (≤ 72 hours after onset, n = 49) was associated with good outcomes in both high-risk (OR = 11.43, 95% confidence interval [CI] = 1.64-127.4, p = 0.01) and medium-risk (OR = 12.80, 95% CI = 2.15-67.8; p = 0.006) ANE-SS categories. No tocilizumab-related adverse events were reported.
INTERPRETATION: Early tocilizumab treatment was associated with improved outcomes in severe ANE, with no adverse events, compared to conventional therapy alone.