{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,6,2]],"date-time":"2026-06-02T09:26:52Z","timestamp":1780392412538,"version":"3.54.1"},"reference-count":17,"publisher":"Springer Science and Business Media LLC","issue":"1","license":[{"start":{"date-parts":[[2019,11,26]],"date-time":"2019-11-26T00:00:00Z","timestamp":1574726400000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"},{"start":{"date-parts":[[2019,11,26]],"date-time":"2019-11-26T00:00:00Z","timestamp":1574726400000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"name":"NCI","award":["K22 CA 234406-01"],"award-info":[{"award-number":["K22 CA 234406-01"]}]}],"content-domain":{"domain":["link.springer.com"],"crossmark-restriction":false},"short-container-title":["J Med Case Reports"],"published-print":{"date-parts":[[2019,12]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:sec>\n<jats:title>Background<\/jats:title>\n<jats:p>Hemophagocytic lymphohistiocytosis is a rare hematological syndrome characterized by excessive and uncontrolled activation of the immune system. The often nonspecific nature of early symptoms and the potential for progression to multiorgan failure and death if appropriate therapy is not started promptly, highlight the importance of heightened recognition for this uncommon disease. Although there are well-described associations of hemophagocytic lymphohistiocytosis with infectious, malignant, and autoimmune diseases and an established treatment protocol for these cases, the link between medications and hemophagocytic lymphohistiocytosis is less clearly established and the optimal treatment of these cases less well defined.<\/jats:p>\n<\/jats:sec><jats:sec>\n<jats:title>Case presentation<\/jats:title>\n<jats:p>Here we describe the case of a 45-year-old caucasian woman presenting with signs and symptoms consistent with hemophagocytic lymphohistiocytosis, induced by recent exposure to lamotrigine. She had a rapidly progressive clinical course, complicated by multiorgan failure including stress-induced Takotsubo cardiomyopathy and cardiac arrest. With dexamethasone and etoposide therapy, she made a full and sustained recovery.<\/jats:p>\n<\/jats:sec><jats:sec>\n<jats:title>Conclusions<\/jats:title>\n<jats:p>This case highlights that medication-induced hemophagocytic lymphohistiocytosis appears to respond similarly to the same dexamethasone and etoposide treatment regimen developed for other non-drug-induced forms of hemophagocytic lymphohistiocytosis. With the continued cessation of the offending agent there has not been need for maintenance therapy and no relapse to date. In addition, given the risk for cardiomyopathy, a clinical complication not classically associated with hemophagocytic lymphohistiocytosis, echocardiogram and telemetry monitoring should be considered in the initial workup of suspected hemophagocytic lymphohistiocytosis.<\/jats:p>\n<\/jats:sec>","DOI":"10.1186\/s13256-019-2295-1","type":"journal-article","created":{"date-parts":[[2019,11,26]],"date-time":"2019-11-26T01:02:29Z","timestamp":1574730149000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":20,"title":["Lamotrigine-induced hemophagocytic lymphohistiocytosis with Takotsubo cardiomyopathy: a case report"],"prefix":"10.1186","volume":"13","author":[{"given":"Jenny Y.","family":"Zhou","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jordan A.","family":"Martinez","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-4588-2775","authenticated-orcid":false,"given":"John Paul","family":"Shen","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2019,11,26]]},"reference":[{"key":"2295_CR1","doi-asserted-by":"publisher","first-page":"69","DOI":"10.2147\/JBM.S46255","volume":"5","author":"MR George","year":"2014","unstructured":"George MR. Hemophagocytic lymphohistiocytosis: review of etiologies and management. J Blood Med. 2014;5:69\u201386.","journal-title":"J Blood Med"},{"issue":"1","key":"2295_CR2","first-page":"31","volume":"49","author":"AT Akenroye","year":"2017","unstructured":"Akenroye AT, Madan N, Mohammadi F, Leider J. Hemophagocytic Lymphohistiocytosis mimics many common conditions: case series and review of literature. Eur Ann Allergy Clin Immunol. 2017;49(1):31\u201341.","journal-title":"Eur Ann Allergy Clin Immunol"},{"issue":"2","key":"2295_CR3","doi-asserted-by":"publisher","first-page":"124","DOI":"10.1002\/pbc.21039","volume":"48","author":"JI Henter","year":"2007","unstructured":"Henter JI, Horne A, Arico M, Egeler RM, Filipovich AH, Imashuku S, et al. HLH-2004: Diagnostic and therapeutic guidelines for hemophagocytic lymphohistiocytosis. Pediatr Blood Cancer. 2007;48(2):124\u201331.","journal-title":"Pediatr Blood Cancer"},{"issue":"4","key":"2295_CR4","doi-asserted-by":"publisher","first-page":"484","DOI":"10.1016\/j.mayocp.2013.12.012","volume":"89","author":"SA Parikh","year":"2014","unstructured":"Parikh SA, Kapoor P, Letendre L, Kumar S, Wolanskyj AP. Prognostic factors and outcomes of adults with hemophagocytic lymphohistiocytosis. Mayo Clin Proc. 2014;89(4):484\u201392.","journal-title":"Mayo Clin Proc"},{"issue":"11","key":"2295_CR5","doi-asserted-by":"publisher","first-page":"1118","DOI":"10.1016\/j.amjmed.2014.04.034","volume":"127","author":"S Riviere","year":"2014","unstructured":"Riviere S, Galicier L, Coppo P, Marzac C, Aumont C, Lambotte O, et al. Reactive hemophagocytic syndrome in adults: a retrospective analysis of 162 patients. Am J Med. 2014;127(11):1118\u201325.","journal-title":"Am J Med"},{"issue":"7","key":"2295_CR6","doi-asserted-by":"publisher","first-page":"592","DOI":"10.1016\/S0002-9343(02)01035-5","volume":"112","author":"O Lambotte","year":"2002","unstructured":"Lambotte O, Costedoat-Chalumeau N, Amoura Z, Piette JC, Cacoub P. Drug-induced hemophagocytosis. Am J Med. 2002;112(7):592\u20133.","journal-title":"Am J Med"},{"issue":"1","key":"2295_CR7","doi-asserted-by":"publisher","first-page":"60","DOI":"10.4103\/2229-5151.152349","volume":"5","author":"D Gauchan","year":"2015","unstructured":"Gauchan D, Shaaban H, Parikh N, Chang NL, Altheeb Z, Maroules M. Severe hemophagocytic lymphohistiocytosis as a complication of drug-induced hypersensitivity syndrome. Int J Crit Illn Inj Sci. 2015;5(1):60\u20131.","journal-title":"Int J Crit Illn Inj Sci"},{"issue":"17","key":"2295_CR8","doi-asserted-by":"publisher","first-page":"4577","DOI":"10.1182\/blood-2011-06-356261","volume":"118","author":"H Trottestam","year":"2011","unstructured":"Trottestam H, Horne A, Arico M, Egeler RM, Filipovich AH, Gadner H, et al. Chemoimmunotherapy for hemophagocytic lymphohistiocytosis: long-term results of the HLH-94 treatment protocol. Blood. 2011;118(17):4577\u201384.","journal-title":"Blood"},{"issue":"15","key":"2295_CR9","doi-asserted-by":"publisher","first-page":"4041","DOI":"10.1182\/blood-2011-03-278127","volume":"118","author":"MB Jordan","year":"2011","unstructured":"Jordan MB, Allen CE, Weitzman S, Filipovich AH, McClain KL. How I treat hemophagocytic lymphohistiocytosis. Blood. 2011;118(15):4041\u201352.","journal-title":"Blood"},{"key":"2295_CR10","volume-title":"FDA Drug Safety Communication: FDA warns of serious immune system reaction with seizure and mental health medicine lamotrigine (Lamictal)","author":"FDA","year":"2018","unstructured":"FDA. FDA Drug Safety Communication: FDA warns of serious immune system reaction with seizure and mental health medicine lamotrigine (Lamictal). In: Administration FD, editor. https:\/\/www.fda.gov\/Drugs\/DrugSafety\/ucm605470.htm. 2018."},{"issue":"4","key":"2295_CR11","doi-asserted-by":"publisher","first-page":"e493","DOI":"10.1097\/MJT.0000000000000515","volume":"24","author":"M Ignaszewski","year":"2017","unstructured":"Ignaszewski M, Ignaszewski MJ, Kohlitz P. Lamotrigine-Associated Hemophagocytic Lymphohistiocytosis. 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A copy of the written consent is available for review by the Editor-in-Chief of this journal.","order":2,"name":"Ethics","group":{"name":"EthicsHeading","label":"Consent for publication"}},{"value":"The authors declare that they have no competing interests.","order":3,"name":"Ethics","group":{"name":"EthicsHeading","label":"Competing interests"}}],"article-number":"345"}}