{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,7,30]],"date-time":"2025-07-30T15:50:40Z","timestamp":1753890640330,"version":"3.41.2"},"reference-count":26,"publisher":"Frontiers Media SA","license":[{"start":{"date-parts":[[2025,5,27]],"date-time":"2025-05-27T00:00:00Z","timestamp":1748304000000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":["frontiersin.org"],"crossmark-restriction":true},"short-container-title":["Front. Cardiovasc. Med."],"abstract":"<jats:sec><jats:title>Background<\/jats:title><jats:p>Acute necrotizing encephalopathy (ANE) is a rare condition characterized by multiple symmetrical brain lesions mainly involving the thalamus. Acute fulminant myocarditis is a diffuse inflammatory disease of the myocardium characterized by acute onset, rapid progression, and a high risk of death. Its pathogenesis involves excessive activation of the innate immune system and the formation of an inflammatory storm. Both conditions are thought to be caused by viral infections. We present a case of ANE with fulminant myocarditis. Reporting this case is important due to the rarity and the critical interplay of these two severe conditions occurring simultaneously.<\/jats:p><\/jats:sec><jats:sec><jats:title>Case presentation<\/jats:title><jats:p>A 16-year-old student presented with a 3-day history of high fever, cough, and expectoration, followed by multiple episodes of convulsive seizures. Despite high doses of vasoactive medications, the patient exhibited low blood pressure and elevated lactate levels. Portable echocardiography revealed diffuse decreased left ventricular motion with severe left ventricular dysfunction (ejection fraction\u2009&amp;lt;\u200930% by visual estimation). The patient was diagnosed with acute fulminant myocarditis. The patient remained comatose with a Glasgow coma scale (GCS) score of 3 (E1VeM1). Brain CT and MRI revealed bilateral striatal, thalamic, and brainstem lesions, typical of ANE. Consequently, a diagnosis of ANE accompanied by fulminant myocarditis was considered. The treatment regimen included high doses of glucocorticoids, immunoglobulins, tocilizumab, and V-A ECMO (Veno-arterial extracorporeal membrane oxygenation) life support. The patient showed significant recovery of cardiac function and was discharged after approximately 24 days of rehabilitation.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusion<\/jats:title><jats:p>This case report highlights the coexistence of ANE and fulminant myocarditis. The underlying mechanisms remain unclear. Early recognition of these two conditions is crucial for prognosis, though challenging. This report underscores the need for heightened awareness and prompt, comprehensive treatment strategies to improve outcomes in such complex cases.<\/jats:p><\/jats:sec>","DOI":"10.3389\/fcvm.2025.1574397","type":"journal-article","created":{"date-parts":[[2025,5,27]],"date-time":"2025-05-27T04:11:43Z","timestamp":1748319103000},"update-policy":"https:\/\/doi.org\/10.3389\/crossmark-policy","source":"Crossref","is-referenced-by-count":0,"title":["A rare case report: acute necrotizing encephalopathy and acute fulminant myocarditis"],"prefix":"10.3389","volume":"12","author":[{"given":"Juan","family":"Ma","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Chenliang","family":"Pan","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Nan","family":"Bai","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Shanshan","family":"Zhang","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Peiling","family":"Mi","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Yongling","family":"Wa","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Andong","family":"Lu","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"1965","published-online":{"date-parts":[[2025,5,27]]},"reference":[{"key":"B1","doi-asserted-by":"publisher","first-page":"555","DOI":"10.1136\/jnnp.58.5.555","article-title":"Acute necrotising encephalopathy of childhood: a new syndrome presenting with multifocal, symmetric brain lesions","volume":"58","author":"Mizuguchi","year":"1995","journal-title":"J Neurol Neurosurg Psychiatry"},{"key":"B2","doi-asserted-by":"publisher","first-page":"259","DOI":"10.1097\/01.inf.0000115631.99896.41","article-title":"Influenza A virus-associated acute necrotizing encephalopathy in the United States","volume":"23","author":"Weitkamp","year":"2004","journal-title":"Pediatr Infect Dis J"},{"key":"B3","doi-asserted-by":"publisher","first-page":"742","DOI":"10.3760\/cma.j.issn.0253-3758.2017.09.004","article-title":"Chinese expert consensus statement on clinical diagnosis and treatment of fulminant myocarditis in adults[J]","volume":"45","year":"2017","journal-title":"Chin J Cardiol"},{"key":"B4","doi-asserted-by":"publisher","first-page":"913","DOI":"10.1007\/s11427-023-2421-0","article-title":"Chinese society of cardiology guidelines on the diagnosis and treatment of adult fulminant myocarditis","volume":"67","author":"Jiang","year":"2024","journal-title":"Sci China Life Sci"},{"key":"B5","doi-asserted-by":"publisher","first-page":"792578","DOI":"10.1155\/2015\/792578","article-title":"Acute necrotizing encephalopathy: an underrecognized clinicoradiologic disorder","volume":"2015","author":"Wu","year":"2015","journal-title":"Mediat Inflamm"},{"key":"B6","doi-asserted-by":"publisher","first-page":"730","DOI":"10.1007\/s12098-023-04598-6","article-title":"Recurrent familial acute necrotizing encephalopathy of childhood (ANEC)","volume":"90","author":"Gowda","year":"2023","journal-title":"Indian J Pediatr"},{"key":"B7","doi-asserted-by":"publisher","first-page":"e17797","DOI":"10.1097\/MD.0000000000017797","article-title":"Use of MRI in the diagnosis and prognosis of acute necrotizing encephalopathy in a Chinese teenager","volume":"98","author":"Li","year":"2019","journal-title":"Medicine (Baltimore)"},{"key":"B8","doi-asserted-by":"publisher","first-page":"ofx145","DOI":"10.1093\/ofid\/ofx145","article-title":"Successful treatment of influenza-associated acute necrotizing encephalitis in an adult using high-dose oseltamivir and methylprednisolone: case report and literature review","volume":"4","author":"Alsolami","year":"2017","journal-title":"Open Forum Infect Dis"},{"key":"B9","doi-asserted-by":"publisher","first-page":"526","DOI":"10.3389\/fped.2020.00526","article-title":"Acute necrotizing encephalopathy of childhood: a multicenter experience in Saudi Arabia","volume":"8","author":"Bashiri","year":"2020","journal-title":"Front Pediatr"},{"key":"B10","doi-asserted-by":"publisher","first-page":"1308044","DOI":"10.3389\/fneur.2023.1308044","article-title":"Clinical characteristics and prognostic analysis of acute necrotizing encephalopathy of childhood: a retrospective study at a single center in China over 3 years","volume":"14","author":"Fang","year":"2023","journal-title":"Front Neurol"},{"key":"B11","doi-asserted-by":"publisher","first-page":"99","DOI":"10.1002\/ped4.12280","article-title":"Plasma exchange therapy for acute necrotizing encephalopathy of childhood","volume":"5","author":"Li","year":"2021","journal-title":"Pediatr Investig"},{"key":"B12","doi-asserted-by":"publisher","first-page":"287","DOI":"10.1038\/s41392-020-00360-y","article-title":"Fulminant myocarditis: a comprehensive review from etiology to treatments and outcomes","volume":"5","author":"Hang","year":"2020","journal-title":"Sig Transduct Target Ther"},{"key":"B13","doi-asserted-by":"publisher","first-page":"670","DOI":"10.1038\/nrcardio.2015.108","article-title":"Viral myocarditis\u2014diagnosis, treatment options and current controversies","volume":"12","author":"Pollack","year":"2015","journal-title":"Nat Rev Cardiol"},{"key":"B14","doi-asserted-by":"publisher","first-page":"2636","DOI":"10.1093\/eurheartj\/eht210","article-title":"Current state of knowledge on aetiology, diagnosis, management, and therapy of myocarditis: a position statement of the European society of cardiology working group on myocardial and pericardial diseases","volume":"34","author":"Caforio","year":"2013","journal-title":"Eur Heart J"},{"key":"B15","doi-asserted-by":"publisher","first-page":"166","DOI":"10.1007\/s11897-014-0196-6","article-title":"Management of fulminant myocarditis: a diagnosis in search of its etiology but with therapeutic options","volume":"11","author":"Maisch","year":"2014","journal-title":"Curr Heart Fail Rep"},{"key":"B16","doi-asserted-by":"publisher","first-page":"114","DOI":"10.31083\/j.rcm2504114","article-title":"Clinical application of extracorporeal membrane oxygenation in the treatment of fulminant myocarditis","volume":"25","author":"Liao","year":"2024","journal-title":"Rev Cardiovasc Med"},{"key":"B17","doi-asserted-by":"publisher","first-page":"1239746","DOI":"10.3389\/fneur.2023.1239746","article-title":"Case report: acute necrotizing encephalopathy: a report of a favorable outcome and systematic meta-analysis of outcomes with different immunosuppressive therapies","volume":"14","author":"Fischell","year":"2023","journal-title":"Front Neurol"},{"key":"B18","doi-asserted-by":"publisher","first-page":"1347885","DOI":"10.3389\/fcvm.2024.1347885","article-title":"A case report of mycoplasma pneumoniae-induced fulminant myocarditis in a 15-year-old male leading to cardiogenic shock and electrical storm","volume":"11","author":"Zhu","year":"2024","journal-title":"Front Cardiovasc Med"},{"key":"B19","doi-asserted-by":"publisher","first-page":"44","DOI":"10.1016\/j.ajhg.2008.12.009","article-title":"Infection-triggered familial or recurrent cases of acute necrotizing encephalopathy caused by mutations in a component of the nuclear pore, RANBP2","volume":"84","author":"Neilson","year":"2009","journal-title":"Am J Human Genet"},{"key":"B20","doi-asserted-by":"publisher","first-page":"2386636","DOI":"10.1080\/07853890.2024.2386636","article-title":"Laboratory diagnosis and treatment of mycoplasma pneumoniae infection in children: a review","volume":"56","author":"Gao","year":"2024","journal-title":"Ann Med"},{"key":"B21","doi-asserted-by":"publisher","first-page":"10","DOI":"10.1128\/jcm.01498-20","article-title":"Evaluation of IgM lateral flow assay as a screening tool for mycoplasma pneumoniae infection in childhood pneumonia","volume":"58","author":"Meyer Sauteur","year":"2020","journal-title":"J Clin Microbiol"},{"key":"B22","doi-asserted-by":"publisher","first-page":"1","DOI":"10.1186\/1471-2180-8-93","article-title":"Polymerase chain reaction is superior to serology for the diagnosis of acute mycoplasma pneumoniae infection and reveals a high rate of persistent infection","volume":"8","author":"Nilsson","year":"2008","journal-title":"BMC Microbiol"},{"key":"B23","doi-asserted-by":"publisher","first-page":"353","DOI":"10.3390\/diagnostics11020353","article-title":"Early confirmation of mycoplasma pneumoniae infection by two short-term serologic IgM examination","volume":"11","author":"Jeon","year":"2021","journal-title":"Diagnostics"},{"key":"B24","first-page":"1331","article-title":"A recovered case of COVID-19 myocarditis and ARDS treated with corticosteroids, tocilizumab, and experimental AT-001","volume":"2","author":"Coyle","year":"2020","journal-title":"Case Rep"},{"key":"B25","doi-asserted-by":"publisher","first-page":"1352","DOI":"10.1093\/rheumatology\/keu019","article-title":"Fulminant myocarditis and macrophage activation syndrome secondary to adult-onset still\u2019s disease successfully treated with tocilizumab","volume":"53","author":"Savage","year":"2014","journal-title":"Rheumatology"},{"key":"B26","doi-asserted-by":"publisher","first-page":"1","DOI":"10.1007\/s00395-014-0449-2","article-title":"Interleukin-6 receptor inhibition modulates the immune reaction and restores titin phosphorylation in experimental myocarditis","volume":"109","author":"Savvatis","year":"2014","journal-title":"Basic Res Cardiol"}],"container-title":["Frontiers in Cardiovascular Medicine"],"original-title":[],"link":[{"URL":"https:\/\/www.frontiersin.org\/articles\/10.3389\/fcvm.2025.1574397\/full","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,5,27]],"date-time":"2025-05-27T04:11:45Z","timestamp":1748319105000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.frontiersin.org\/articles\/10.3389\/fcvm.2025.1574397\/full"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2025,5,27]]},"references-count":26,"alternative-id":["10.3389\/fcvm.2025.1574397"],"URL":"https:\/\/doi.org\/10.3389\/fcvm.2025.1574397","relation":{},"ISSN":["2297-055X"],"issn-type":[{"type":"electronic","value":"2297-055X"}],"subject":[],"published":{"date-parts":[[2025,5,27]]},"article-number":"1574397"}}