{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,26]],"date-time":"2026-08-26T16:01:48Z","timestamp":1787760108222,"version":"build-2784847793"},"reference-count":19,"publisher":"Wiley","issue":"9","license":[{"start":{"date-parts":[[2023,2,7]],"date-time":"2023-02-07T00:00:00Z","timestamp":1675728000000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"funder":[{"DOI":"10.13039\/100009647","name":"Ministry of Health","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100009647","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100000764","name":"King's College London","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100000764","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Develop Med Child Neuro"],"published-print":{"date-parts":[[2023,9]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:p>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\u00a0modified Rankin Scale) in Malaysia between February 2019 and January 2020. Twenty\u2010seven children (12 male), aged 7\u2009months to 14\u2009years (mean 4\u2009years) 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\u2009+\u2009intravenous immunoglobulin [IVIG]) and 16 did not (two received MTP alone, 14 received MTP\u2009+\u2009IVIG). Nine died. Among the survivors, six had good outcomes (modified Rankin Score 0\u20132) at 6\u2009months follow\u2010up. All patients who received tocilizumab in combination with MTP\u2009+\u2009IVIG survived. Twenty children received first immunotherapy within 48\u2009hours of admission. No significant association was found between the timing of first immunotherapy with outcomes. Those with brainstem dysfunction (<jats:italic>p<\/jats:italic>\u00a0=\u20090.016) 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\u2009+\u2009IVIG. However, larger studies will be needed to determine the effect of this regime on the long\u2010term outcomes.<\/jats:p>","DOI":"10.1111\/dmcn.15536","type":"journal-article","created":{"date-parts":[[2023,2,7]],"date-time":"2023-02-07T09:53:10Z","timestamp":1675763590000},"page":"1256-1263","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":27,"title":["Factors associated with outcomes of severe acute necrotizing encephalopathy: A multicentre experience in Malaysia"],"prefix":"10.1111","volume":"65","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-3988-204X","authenticated-orcid":false,"given":"Vanessa Wan Mun","family":"Lee","sequence":"first","affiliation":[{"name":"Paediatric Neurology Unit Hospital Tunku Azizah  Kuala Lumpur Malaysia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Teik Beng","family":"Khoo","sequence":"additional","affiliation":[{"name":"Paediatric Neurology Unit Hospital Tunku Azizah  Kuala Lumpur Malaysia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Chee Ming","family":"Teh","sequence":"additional","affiliation":[{"name":"Pediatric Neurology Unit Hospital Pulau Pinang  Pulau Pinang Malaysia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hock Sin","family":"Heng","sequence":"additional","affiliation":[{"name":"Pediatric Neurology Unit Sabah Women and Children Hospital  Kota Kinabalu Malaysia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Limin","family":"Li","sequence":"additional","affiliation":[{"name":"Division of Pediatric Neurology, Department of Paediatrics, Faculty of Medicine University Malaya  Kuala Lumpur Malaysia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yusma Lyana Md","family":"Yusof","sequence":"additional","affiliation":[{"name":"Pediatric Unit, Faculty of Medicine Universiti Teknologi Mara  Selangor Darul Ehsan Malaysia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Nor Azni","family":"Yahaya","sequence":"additional","affiliation":[{"name":"Paediatric Neurology Unit Hospital Raja Perempuan Zainab II  Kota Bharu Malaysia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sangita","family":"Dharshini","sequence":"additional","affiliation":[{"name":"Pediatric Neurology Unit Subang Jaya Medical Centre  Selangor Darul Ehsan Malaysia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sau Wei","family":"Wong","sequence":"additional","affiliation":[{"name":"Paediatric Neurology Unit Hospital Pakar Kanak\u2010Kanak Universiti Kebangsaan Malaysia  Kuala Lumpur Malaysia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Tai","family":"Nickson","sequence":"additional","affiliation":[{"name":"Paediatric Unit Hospital Umum Sarawak  Sarawak Malaysia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"name":"ANE Malaysia Outcome Study 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