Acute Necrotizing Encephalopathy in Children: Insights and Outcomes from Iran A Single Centre Original Research in Acute Necrotizing Encephalopathy of Childhood
Iranian Journal of Child Neurology,
Vol. 18 No. 4 (2024),
29 September 2024
,
Page 107-119
https://doi.org/10.22037/ijcn.v18i4.45458
Abstract
Objectives
Acute necrotizing encephalopathy of childhood (ANEC) is a rare, potentially life-threatening condition. This study aimed to identify clinical profiles and outcomes of ANEC while assessing the accuracy of severity scoring in the Iranian population.
Materials & Methods
The present study collected demographic, clinical, laboratory, and radiological data from children diagnosed with ANEC. Severity was measured using the ANE-Severity Score (ANE-SS), while outcomes were assessed with the Glasgow Outcome Score (GOS). This research analyzed the relationship between these scores and various parameters for statistical significance.
Results
Seven patients were included over three years, with an average age of 4.4±2.7 years (5 males). ANE-SS varied from moderate to high, with most patients experiencing moderate to severe disabilities, as indicated by the GOS. Significant correlations were found with initial serum magnesium levels, pupil light reactivity, and initial GCS score (P-value < 0.05).
Conclusion
Controlling initial magnesium levels may improve ANEC outcomes. Additionally, intact pupil light reactivity at admission was associated with a better prognosis.
- Acute Necrotizing Encephalopathy
- Magnesium
- Pediatric Neurology
- Hypernatremia
- Epidemiology
How to Cite
References
Mizuguchi M. Acute necrotizing encephalopathy of childhood: a novel form of acute encephalopathy prevalent in Japan and Taiwan. Brain Dev. 1997;19(2):81-92. doi:10.1016/s0387-7604(96)00063-0
Jan F, Jafri SK, Ibrahim SH. Acute Necrotizing Encephalopathy. J Coll Physicians Surg Pak. 2019;29(7):649-653. doi:10.29271/jcpsp.2019.07.649
Bashiri FA, Al Johani S, Hamad MH, et al. Acute Necrotizing Encephalopathy of Childhood: A Multicenter Experience in Saudi Arabia . Front Pediatr . 2020;8. https://www.frontiersin.org/articles/10.3389/fped.2020.00526
Ibrahim RSM, Elzayat W, Seif HM, et al. Multi-parametric magnetic resonance imaging in acute necrotizing encephalopathy of children: validity and prognostic value. Egypt J Radiol Nucl Med. 2020;51(1):113. doi:10.1186/s43055-020-00214-1
Salehiomran MR, Nooreddini H, Baghdadi F. Acute necrotizing encephalopathy of childhood; a case report. Iran J child Neurol. 2013;7(2):51-54.
Gupta S, Banerjee B, Sasidharan SK, Acharya U V. Clinical, laboratory, radiologic profile, and outcome in acute necrotizing encephalopathy of childhood (ANEC) – A case series. J Pediatr Crit Care. 2021;8(4). https://journals.lww.com/jpcr/fulltext/2021/08040/clinical,_laboratory,_radiologic_profile,_and.7.aspx
Fang Y, Gao Q, Jin W, et al. Clinical characteristics and prognostic analysis of acute necrotizing encephalopathy of childhood: a retrospective study at a single center in China over 3 years. Front Neurol. 2023;14. https://www.frontiersin.org/journals/neurology/articles/10.3389/fneur.2023.1308044
Vanjare HA, Selvi BT, Karuppusami R, et al. Clinical and Radiologic Findings of Acute Necrotizing Encephalopathy in Young Adults. AJNR Am J Neuroradiol. 2020;41(12):2250-2254. doi:10.3174/ajnr.A6803
Yamamoto H, Okumura A, Natsume J, Kojima S, Mizuguchi M. A severity score for acute necrotizing encephalopathy. Brain Dev. 2015;37(3):322-327. doi:10.1016/j.braindev.2014.05.007
McMillan T, Wilson L, Ponsford J, Levin H, Teasdale G, Bond M. The Glasgow Outcome Scale — 40 years of application and refinement. Nat Rev Neurol. 2016;12(8):477-485. doi:10.1038/nrneurol.2016.89
Ortiz JF, Ruxmohan S, Saxena A, et al. Minocycline and Magnesium As Neuroprotective Agents for Ischemic Stroke: A Systematic Review. Cureus. 2020;12(12):e12339. doi:10.7759/cureus.12339
Maier JAM, Locatelli L, Fedele G, Cazzaniga A, Mazur A. Magnesium and the Brain: A Focus on Neuroinflammation and Neurodegeneration. Int J Mol Sci. 2022;24(1). doi:10.3390/ijms24010223
Yamanaka R, Shindo Y, Oka K. Magnesium Is a Key Player in Neuronal Maturation and Neuropathology. Int J Mol Sci. 2019;20(14). doi:10.3390/ijms20143439
Kuwamura M, Inumaki K, Tanaka M, et al. Oligodendroglial pathology in the development of myelin breakdown in the dmy mutant rat. Brain Res. 2011;1389:161-168. doi:10.1016/j.brainres.2011.03.009
Kuramoto T, Kuwamura M, Tokuda S, et al. A mutation in the gene encoding mitochondrial Mg2+ channel MRS2 results in demyelination in the rat. PLoS Genet. 2011;7(1):e1001262. doi:10.1371/journal.pgen.1001262
Itoh K, Maki T, Shindo A, et al. Magnesium sulfate protects oligodendrocyte lineage cells in a rat cell-culture model of hypoxic-ischemic injury. Neurosci Res. 2016;106:66-69. doi:10.1016/j.neures.2015.12.004
Lingam I, Robertson NJ. Magnesium as a Neuroprotective Agent: A Review of Its Use in the Fetus, Term Infant with Neonatal Encephalopathy, and the Adult Stroke Patient. Dev Neurosci. 2018;40(1):1-12. doi:10.1159/000484891
Kirkland AE, Sarlo GL, Holton KF. The Role of Magnesium in Neurological Disorders. Nutrients. 2018;10(6). doi:10.3390/nu10060730
Wu H, Bai M, Li X, Xing Y, Sun S. Diagnosis and treatment of brain injury complicated by hypernatremia. Front Neurol. 2022;13. https://www.frontiersin.org/journals/neurology/articles/10.3389/fneur.2022.1026540
Feigin E, Feigin L, Ingbir M, Ben-Bassat OK, Shepshelovich D. Rate of Correction and All-Cause Mortality in Patients With Severe Hypernatremia. JAMA Netw Open. 2023;6(9):e2335415-e2335415. doi:10.1001/jamanetworkopen.2023.35415
Sterns RH. Disorders of plasma sodium--causes, consequences, and correction. N Engl J Med. 2015;372(1):55-65. doi:10.1056/NEJMra1404489
Sterns RH. Evidence for Managing Hypernatremia: Is It Just Hyponatremia in Reverse? Clin J Am Soc Nephrol. 2019;14(5):645-647. doi:10.2215/CJN.02950319
Didsbury M, See EJ, Cheng DR, Kausman J, Quinlan C. Correcting Hypernatremia in Children. Clin J Am Soc Nephrol. 2023;18(3):306-314. doi:10.2215/CJN.0000000000000077
Wu L, Peng H, Jiang Y, He L, Jiang L, Hu Y. Clinical features and imaging manifestations of acute necrotizing encephalopathy in children. Int J Dev Neurosci Off J Int Soc Dev Neurosci. 2022;82(5):447-457. doi:10.1002/jdn.10201
Nass RD, Zur B, Elger CE, Holdenrieder S, Surges R. Acute metabolic effects of tonic-clonic seizures. Epilepsia open. 2019;4(4):599-608. doi:10.1002/epi4.12364
Riemann A, Ihling A, Thomas J, Schneider B, Thews O, Gekle M. Acidic environment activates inflammatory programs in fibroblasts via a cAMP–MAPK pathway. Biochim Biophys Acta - Mol Cell Res. 2015;1853(2):299-307. doi:https://doi.org/10.1016/j.bbamcr.2014.11.022
Majdan M, Steyerberg EW, Nieboer D, Mauritz W, Rusnak M, Lingsma HF. Glasgow coma scale motor score and pupillary reaction to predict six-month mortality in patients with traumatic brain injury: comparison of field and admission assessment. J Neurotrauma. 2015;32(2):101-108. doi:10.1089/neu.2014.3438
Javaudin F, Leclere B, Segard J, et al. Prognostic performance of early absence of pupillary light reaction after recovery of out of hospital cardiac arrest. Resuscitation. 2018;127:8-13. doi:10.1016/j.resuscitation.2018.03.020
Abend NS, Licht DJ. Predicting outcome in children with hypoxic ischemic encephalopathy. Pediatr Crit Care Med. 2008;9(1). https://journals.lww.com/pccmjournal/fulltext/2008/01000/predicting_outcome_in_children_with_hypoxic.7.aspx
Fong CY, Saw MT, Li L, Lim WK, Ong LC, Gan CS. Malaysian outcome of acute necrotising encephalopathy of childhood. Brain Dev. 2021;43(4):538-547. doi:https://doi.org/10.1016/j.braindev.2020.12.003
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