{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,3]],"date-time":"2026-07-03T21:27:25Z","timestamp":1783114045314,"version":"3.54.6"},"reference-count":15,"publisher":"Oxford University Press (OUP)","issue":"6","license":[{"start":{"date-parts":[[2021,6,16]],"date-time":"2021-06-16T00:00:00Z","timestamp":1623801600000},"content-version":"vor","delay-in-days":18,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2021,5,29]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:sec><jats:title>Background<\/jats:title><jats:p>Fulminant myocarditis is a life-threatening condition characterized by acute cardiac dysfunction requiring pharmacological or mechanical circulatory support. Haemophagocytic lymphohistiocytosis (HLH) is an uncommon state of immune dysregulation and overactivation. Inflammation mediated by interleukin-1 (IL-1) is thought to play a role in the pathogenesis of myocarditis and HLH, and there is some evidence that the IL-1 receptor antagonist Anakinra may play a role in treating both these conditions.<\/jats:p><\/jats:sec><jats:sec><jats:title>Case summary<\/jats:title><jats:p>A 26-year-old previously healthy male presented to the Emergency Department with a 3-day history of malaise, headache, vomiting, diarrhoea, and fever. He was profoundly hypotensive on arrival, diagnosed with septic shock, and commenced on broad-spectrum antibiotics and vasopressors. Blood tests showed lymphopenia, thrombocytopenia, low fibrinogen and elevated high sensitivity troponin T, ferritin, and C-reactive protein. Echocardiography demonstrated severely impaired biventricular systolic function and a diagnosis of fulminant myocarditis was made. His condition deteriorated and he required intubation and additional inotropic support. A diagnosis of HLH was made and he was commenced on Anakinra and Methylprednisolone. His condition improved rapidly thereafter. Polymerase chain reaction testing subsequently confirmed infection with Neisseria meningitidis.<\/jats:p><\/jats:sec><jats:sec><jats:title>Discussion<\/jats:title><jats:p>In this case, fulminant myocarditis and HLH were life-threatening manifestations of meningococcal septicaemia, and the patient\u2019s condition improved rapidly following administration of the IL-1 receptor antagonist Anakinra. These complications should be borne in mind in septic patients with marked haemodynamic instability and multiorgan dysfunction, and treatment with Anakinra should be considered in those who fail to respond to conventional therapy.<\/jats:p><\/jats:sec>","DOI":"10.1093\/ehjcr\/ytab201","type":"journal-article","created":{"date-parts":[[2021,6,16]],"date-time":"2021-06-16T04:48:53Z","timestamp":1623818933000},"source":"Crossref","is-referenced-by-count":9,"title":["Anakinra treats fulminant myocarditis from<i>Neisseria meningitidis<\/i>septicaemia and haemophagocytic lymphohistiocytosis: a case report"],"prefix":"10.1093","volume":"5","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-8229-8060","authenticated-orcid":false,"given":"Ross J","family":"Thomson","sequence":"first","affiliation":[{"name":"Centre for Inherited Heart Muscle Conditions, Department of Cardiology, Royal Free London NHS Foundation Trust, Pond Street, London NW3 2QG, UK"},{"name":"William Harvey Research Institute, Queen Mary University of London, Charthouse Square, London EC1M 6BQ, UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Animesh","family":"Singh","sequence":"additional","affiliation":[{"name":"Department of Rheumatology, Royal Free London NHS Foundation Trust, Pond Street, London NW3 2QG, UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Daniel S","family":"Knight","sequence":"additional","affiliation":[{"name":"UCL Institute of Cardiovascular Science, University College London, Gower Street, London WC1E 6BT, UK"},{"name":"Cardiovascular Magnetic Resonance Unit, The Royal Free London NHS Foundation Trust, Pond Street, London NW3 2QG, UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-8110-5135","authenticated-orcid":false,"given":"Jim","family":"Buckley","sequence":"additional","affiliation":[{"name":"Department of Intensive Care Medicine, Royal Free London NHS Foundation Trust, Pond Street, London NW3 2QG, UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lucy E","family":"Lamb","sequence":"additional","affiliation":[{"name":"Department of Infectious Diseases, Royal Free London NHS Foundation Trust, Pond Street, London NW3 2QG, UK"},{"name":"Academic Department of Military Medicine, Royal Centre for Defence Medicine, Birmingham, UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-5662-0642","authenticated-orcid":false,"given":"Gabriella","family":"Captur","sequence":"additional","affiliation":[{"name":"Centre for Inherited Heart Muscle Conditions, Department of Cardiology, Royal Free London NHS Foundation Trust, Pond Street, London NW3 2QG, UK"},{"name":"UCL Institute of Cardiovascular Science, University College London, Gower Street, London WC1E 6BT, UK"},{"name":"MRC Unit for Lifelong Health and Ageing at UCL, 1-19 Torrington Place, London WC1E 7HB, UK"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"286","published-online":{"date-parts":[[2021,6,16]]},"reference":[{"key":"2021070816595991000_ytab201-B1","doi-asserted-by":"crossref","first-page":"114","DOI":"10.1007\/s11886-018-1054-z","article-title":"Acute and fulminant myocarditis: a pragmatic clinical approach to diagnosis and treatment","volume":"20","author":"Ammirati","year":"2018","journal-title":"Curr Cardiol Rep"},{"key":"2021070816595991000_ytab201-B2","doi-asserted-by":"crossref","first-page":"693","DOI":"10.1038\/ncpcardio1331","article-title":"Fulminant myocarditis","volume":"5","author":"Gupta","year":"2008","journal-title":"Nat Clin Pract Cardiovasc Med"},{"key":"2021070816595991000_ytab201-B3","doi-asserted-by":"crossref","first-page":"220","DOI":"10.1016\/j.ijcard.2015.04.071","article-title":"Myocarditis in a young man with adult onset Still\u2019s disease successfully treated with Il-1 blocker","volume":"189","author":"Luconi","year":"2015","journal-title":"Int J Cardiol"},{"key":"2021070816595991000_ytab201-B4","doi-asserted-by":"crossref","first-page":"119","DOI":"10.3389\/fimmu.2019.00119","article-title":"The immunology of macrophage activation syndrome","volume":"10","author":"Crayne","year":"2019","journal-title":"Front Immunol"},{"key":"2021070816595991000_ytab201-B5","doi-asserted-by":"crossref","first-page":"417","DOI":"10.1093\/rheumatology\/keq218","article-title":"Successful treatment of severe paediatric rheumatic disease-associated macrophage activation syndrome with interleukin-1 inhibition following conventional immunosuppressive therapy: case series with 12 patients","volume":"50","author":"Miettunen","year":"2011","journal-title":"Rheumatology"},{"key":"2021070816595991000_ytab201-B6","doi-asserted-by":"crossref","first-page":"2613","DOI":"10.1002\/art.38690","article-title":"Development and validation of the HScore, a score for the diagnosis of reactive hemophagocytic syndrome","volume":"66","author":"Fardet","year":"2014","journal-title":"Arthritis Rheumatol"},{"key":"2021070816595991000_ytab201-B7","doi-asserted-by":"crossref","first-page":"283","DOI":"10.1046\/j.1365-2796.2002.01019.x","article-title":"Inflammatory bio-markers and cardiovascular risk prediction","volume":"252","author":"Blake","year":"2002","journal-title":"J Intern Med"},{"key":"2021070816595991000_ytab201-B8","doi-asserted-by":"crossref","first-page":"113","DOI":"10.1093\/eurheartj\/ehaa099","article-title":"Targeting cardiovascular inflammation: next steps in clinical translation","volume":"42","author":"Lawler","year":"2021","journal-title":"Eur Heart J"},{"key":"2021070816595991000_ytab201-B9","doi-asserted-by":"crossref","first-page":"96","DOI":"10.1161\/01.CIR.100.1.96","article-title":"C-Reactive protein as a cardiovascular risk factor","volume":"100","author":"Lagrand","year":"1999","journal-title":"Circulation"},{"key":"2021070816595991000_ytab201-B10","doi-asserted-by":"crossref","first-page":"1119","DOI":"10.1056\/NEJMoa1707914","article-title":"Antiinflammatory therapy with canakinumab for atherosclerotic disease","volume":"377","author":"Ridker","year":"2017","journal-title":"N Engl J Med"},{"key":"2021070816595991000_ytab201-B11","doi-asserted-by":"crossref","first-page":"e33438","DOI":"10.1371\/journal.pone.0033438","article-title":"Enhanced interleukin-1 activity contributes to exercise intolerance in patients with systolic heart failure","volume":"7","author":"van Tassell","year":"2012","journal-title":"PLos One"},{"key":"2021070816595991000_ytab201-B12","doi-asserted-by":"crossref","first-page":"949","DOI":"10.1084\/jem.183.3.949","article-title":"Tumor necrosis factor alpha and interleukin 1beta are responsible for in vitro myocardial cell depression induced by human septic shock serum","volume":"183","author":"Kumar","year":"1996","journal-title":"J Exp Med"},{"key":"2021070816595991000_ytab201-B13","doi-asserted-by":"crossref","first-page":"e004373","DOI":"10.1161\/CIRCHEARTFAILURE.117.004373","article-title":"Interleukin-1 blockade in recently decompensated systolic heart failure","volume":"10","author":"Tassell","year":"2017","journal-title":"Circulation Hear Fail"},{"key":"2021070816595991000_ytab201-B14","doi-asserted-by":"crossref","first-page":"1995","DOI":"10.1093\/eurheartj\/ehp249","article-title":"Randomized study on the efficacy of immunosuppressive therapy in patients with virus-negative inflammatory cardiomyopathy: the TIMIC study","volume":"30","author":"Frustaci","year":"2009","journal-title":"Eur Heart J"},{"key":"2021070816595991000_ytab201-B15","doi-asserted-by":"crossref","first-page":"3329","DOI":"10.1007\/s10067-018-4095-1","article-title":"Anakinra treatment in macrophage activation syndrome: a single center experience and systemic review of literature","volume":"37","author":"S\u00f6nmez","year":"2018","journal-title":"Clin Rheumatol"}],"container-title":["European Heart Journal - Case Reports"],"original-title":[],"language":"en","link":[{"URL":"http:\/\/academic.oup.com\/ehjcr\/article-pdf\/5\/6\/ytab201\/38656500\/ytab201.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"syndication"},{"URL":"http:\/\/academic.oup.com\/ehjcr\/article-pdf\/5\/6\/ytab201\/38656500\/ytab201.pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2022,12,31]],"date-time":"2022-12-31T08:22:37Z","timestamp":1672474957000},"score":1,"resource":{"primary":{"URL":"https:\/\/academic.oup.com\/ehjcr\/article\/doi\/10.1093\/ehjcr\/ytab201\/6300013"}},"subtitle":[],"editor":[{"given":"Ching-Hui","family":"Sia","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"editor"}]},{"given":"Andriana","family":"Anagnostopoulou","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"editor"}]},{"given":"Filipe Penna De","family":"Carvalho","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"editor"}]},{"given":"Mark","family":"Abela","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"editor"}]},{"given":"Bretty Sydney","family":"Bernstein","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"editor"}]},{"given":"Christian Fielder","family":"Camm","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"editor"}]}],"short-title":[],"issued":{"date-parts":[[2021,5,29]]},"references-count":15,"journal-issue":{"issue":"6","published-print":{"date-parts":[[2021,5,29]]}},"URL":"https:\/\/doi.org\/10.1093\/ehjcr\/ytab201","relation":{},"ISSN":["2514-2119"],"issn-type":[{"value":"2514-2119","type":"electronic"}],"subject":[],"published-other":{"date-parts":[[2021,6,1]]},"published":{"date-parts":[[2021,5,29]]},"article-number":"ytab201"}}