{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,9]],"date-time":"2026-07-09T22:45:31Z","timestamp":1783637131859,"version":"3.55.0"},"reference-count":8,"publisher":"BMJ","issue":"12","content-domain":{"domain":["bmj.com"],"crossmark-restriction":true},"short-container-title":["BMJ Case Rep"],"accepted":{"date-parts":[[2021,11,23]]},"published-print":{"date-parts":[[2021,12]]},"abstract":"<jats:p>We describe two young cases of reactive haemophagocytic lymphohistiocytosis (HLH) with the resultant stress cardiomyopathy in the setting of underlying autoimmune diseases, systemic lupus erythematosus (SLE) and Still\u2019s disease. The initial presentation was similar in both cases with fever, hyperinflammatory response, hypotension (vasoplegia), bicytopenia and hyperferritinemia. Despite standard of care and multiple broad-spectrum antibiotics, both cases remained pyrexic and were ultimately admitted to the intensive therapy unit to treat cardiogenic shock. Echocardiogram of both cases showed low ejection fraction, the cause for which was not found until the final diagnosis of HLH was made. Both cases made a complete clinical and cardiac recovery following the initiation of high-dose glucocorticoids and anakinra.<\/jats:p>","DOI":"10.1136\/bcr-2021-246416","type":"journal-article","created":{"date-parts":[[2021,12,9]],"date-time":"2021-12-09T11:35:49Z","timestamp":1639049749000},"page":"e246416","update-policy":"https:\/\/doi.org\/10.1136\/crossmarkpolicy","source":"Crossref","is-referenced-by-count":6,"title":["Haemophagocytic lymphohistiocytosis (HLH)-associated stress cardiomyopathy secondary to autoimmune conditions successfully treated with anakinra"],"prefix":"10.1136","volume":"14","author":[{"given":"Muhammad","family":"Khalid","sequence":"first","affiliation":[{"name":"Rheumatology Department, Mater Misericordiae University Hospital, Dublin 7, Ireland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-5596-4726","authenticated-orcid":false,"given":"Abuelmagd","family":"Abdalla","sequence":"additional","affiliation":[{"name":"Rheumatology Department, Mater Misericordiae University Hospital, Dublin 7, Ireland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Geraldine","family":"McCarthy","sequence":"additional","affiliation":[{"name":"Rheumatology Department, Mater Misericordiae University Hospital, Dublin 7, Ireland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"John","family":"Stack","sequence":"additional","affiliation":[{"name":"Rheumatology Department, Mater Misericordiae University Hospital, Dublin 7, Ireland"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"239","published-online":{"date-parts":[[2021,12,9]]},"reference":[{"key":"2025082712384223000_14.12.e246416.1","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":"2025082712384223000_14.12.e246416.2","doi-asserted-by":"publisher","DOI":"10.1146\/annurev-med-041610-134208"},{"key":"2025082712384223000_14.12.e246416.3","doi-asserted-by":"crossref","first-page":"1","DOI":"10.1186\/s13054-016-1369-3","article-title":"Your critical care patient may have HLH (hemophagocytic lymphohistiocytosis)","volume":"20","author":"Machowicz","year":"2016","journal-title":"Crit Care"},{"key":"2025082712384223000_14.12.e246416.4","doi-asserted-by":"publisher","DOI":"10.1182\/blood.2018894618"},{"key":"2025082712384223000_14.12.e246416.5","doi-asserted-by":"publisher","DOI":"10.1002\/pbc.21039"},{"key":"2025082712384223000_14.12.e246416.6","doi-asserted-by":"crossref","DOI":"10.1016\/j.chest.2016.08.448","article-title":"Takotsubo cardiomyopathy due to hemophagocytic lymphohistiocytosis","volume":"150","author":"Abdin","year":"2016","journal-title":"Chest"},{"key":"2025082712384223000_14.12.e246416.7","doi-asserted-by":"crossref","first-page":"205","DOI":"10.1159\/000098461","article-title":"Two cases of ampulla (takotsubo-shaped) cardiomyopathy associated with hemophagocytic lymphohistiocytosis","volume":"117","author":"Takeoka","year":"2007","journal-title":"Acta Haematol"},{"key":"2025082712384223000_14.12.e246416.8","doi-asserted-by":"crossref","DOI":"10.1136\/bcr-2018-226599","article-title":"Takotsubo cardiomyopathy secondary to haemophagocytic lymphohistiocytosis in HIV patients: a comprehensive review","volume":"12","author":"Ullah","year":"2019","journal-title":"BMJ Case Rep"}],"container-title":["BMJ Case Reports"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/syndication.highwire.org\/content\/doi\/10.1136\/bcr-2021-246416","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,8,27]],"date-time":"2025-08-27T19:38:50Z","timestamp":1756323530000},"score":1,"resource":{"primary":{"URL":"https:\/\/casereports.bmj.com\/lookup\/doi\/10.1136\/bcr-2021-246416"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2021,12]]},"references-count":8,"journal-issue":{"issue":"12","published-online":{"date-parts":[[2021,12,9]]},"published-print":{"date-parts":[[2021,12]]}},"alternative-id":["10.1136\/bcr-2021-246416"],"URL":"https:\/\/doi.org\/10.1136\/bcr-2021-246416","relation":{},"ISSN":["1757-790X"],"issn-type":[{"value":"1757-790X","type":"electronic"}],"subject":[],"published":{"date-parts":[[2021,12]]}}}