{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,6,17]],"date-time":"2026-06-17T21:43:07Z","timestamp":1781732587719,"version":"3.54.5"},"reference-count":24,"publisher":"Oxford University Press (OUP)","issue":"5","content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2021,9,30]]},"abstract":"<jats:title>Abstract<\/jats:title>\n               <jats:p>This case report describes a case of fatal toxic epidermal necrolysis complicated by both vanishing bile duct syndrome and hemophagocytic lymphohistiocytosis due to Influenza B infection. Here we highlight the potential for complex morbidity secondary to underlying autoimmune hypersensitivity. Furthermore, the stepwise progression of these pathologies is noted, with the initial epidermal lesions first progressing to cholestatic injury and then subsequently to the hematologic manifestations.<\/jats:p>","DOI":"10.1093\/jbcr\/irab058","type":"journal-article","created":{"date-parts":[[2021,4,27]],"date-time":"2021-04-27T14:17:11Z","timestamp":1619533031000},"page":"1043-1046","source":"Crossref","is-referenced-by-count":3,"title":["A Fatal Case of Toxic Epidermal Necrolysis Combined With Vanishing Bile Duct Syndrome and Hemophagocytic Lymphohistiocytosis"],"prefix":"10.1093","volume":"42","author":[{"given":"Julian","family":"Wier","sequence":"first","affiliation":[{"name":"Keck School of Medicine, University of Southern California, Los Angeles, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Alexandra","family":"Lacey","sequence":"additional","affiliation":[{"name":"Keck School of Medicine, University of Southern California, Los Angeles, USA"},{"name":"Division of Plastic and Reconstructive Surgery, Keck School of Medicine, University of Southern California, Los Angeles, USA"},{"name":"Los Angeles County Regional Burn Center, Los Angeles County + University of Southern California Medical Center, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Haig","family":"Yenikomshian","sequence":"additional","affiliation":[{"name":"Keck School of Medicine, University of Southern California, Los Angeles, USA"},{"name":"Division of Plastic and Reconstructive Surgery, Keck School of Medicine, University of Southern California, Los Angeles, USA"},{"name":"Los Angeles County Regional Burn Center, Los Angeles County + University of Southern California Medical Center, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Justin","family":"Gillenwater","sequence":"additional","affiliation":[{"name":"Keck School of Medicine, University of Southern California, Los Angeles, USA"},{"name":"Division of Plastic and Reconstructive Surgery, Keck School of Medicine, University of Southern California, Los Angeles, USA"},{"name":"Los Angeles County Regional Burn Center, Los Angeles County + University of Southern California Medical Center, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"286","published-online":{"date-parts":[[2021,4,27]]},"reference":[{"key":"2021093015370270600_CIT0001","doi-asserted-by":"crossref","first-page":"219","DOI":"10.1016\/j.jaci.2015.05.050","article-title":"Evolving models of the immunopathogenesis of T cell-mediated drug allergy: the role of host, pathogens, and drug response","volume":"136","author":"White","year":"2015","journal-title":"J Allergy Clin Immunol"},{"key":"2021093015370270600_CIT0002","doi-asserted-by":"crossref","first-page":"6431694","DOI":"10.1155\/2018\/6431694","article-title":"An updated review of the molecular mechanisms in drug hypersensitivity","volume":"2018","author":"Chen","year":"2018","journal-title":"J Immunol Res"},{"key":"2021093015370270600_CIT0003","doi-asserted-by":"crossref","first-page":"149","DOI":"10.1016\/j.trim.2010.06.008","article-title":"Alloreactivity from human viral specific memory T-cells","volume":"23","author":"D\u2019Orsogna","year":"2010","journal-title":"Transpl Immunol"},{"key":"2021093015370270600_CIT0004","doi-asserted-by":"crossref","first-page":"e45","DOI":"10.1111\/pde.13370","article-title":"Influenza B virus infection and Stevens\u2013Johnson syndrome","volume":"35","author":"Tamez","year":"2018","journal-title":"Pediatr Dermatol"},{"key":"2021093015370270600_CIT0005","doi-asserted-by":"crossref","first-page":"929","DOI":"10.1111\/pde.13921","article-title":"Two pediatric cases of influenza B-induced rash and mucositis: Stevens\u2013Johnson syndrome or expansion of the Mycoplasma pneumoniae-induced rash with mucositis (MIRM) spectrum?","volume":"36","author":"Goyal","year":"2019","journal-title":"Pediatr Dermatol"},{"key":"2021093015370270600_CIT0006","doi-asserted-by":"crossref","first-page":"1387","DOI":"10.1016\/j.jid.2016.03.023","article-title":"Morbidity and mortality of Stevens\u2013Johnson syndrome and toxic epidermal necrolysis in United States adults","volume":"136","author":"Hsu","year":"2016","journal-title":"J Invest Dermatol"},{"key":"2021093015370270600_CIT0007","first-page":"5738","article-title":"The ambiguous boundary between EBV-related hemophagocytic lymphohistiocytosis and systemic EBV-driven T cell lymphoproliferative disorder","volume":"7","author":"Smith","year":"2014","journal-title":"Int J Clin Exp Pathol"},{"key":"2021093015370270600_CIT0008","doi-asserted-by":"crossref","first-page":"412","DOI":"10.1111\/bjh.13837","article-title":"Haemophagocytic lymphohistiocytosis in adults: a multicentre case series over 7 years","volume":"172","author":"Schram","year":"2016","journal-title":"Br J Haematol"},{"key":"2021093015370270600_CIT0009","doi-asserted-by":"crossref","first-page":"470","DOI":"10.1007\/BF02984011","article-title":"Serum granulysin is elevated in patients with hemophagocytic lymphohistiocytosis","volume":"86","author":"Nagasawa","year":"2007","journal-title":"Int J Hematol"},{"key":"2021093015370270600_CIT0010","doi-asserted-by":"crossref","first-page":"18","DOI":"10.1016\/j.clim.2019.03.006","article-title":"The inhibitory receptors on NK cells and CTLs are upregulated in adult and adolescent patients with secondary hemophagocytic lymphohistiocytosis","volume":"202","author":"Gao","year":"2019","journal-title":"Clin Immunol"},{"key":"2021093015370270600_CIT0011","first-page":"125","article-title":"What\u2019s beneath the surface? Stevens\u2013Johnson syndrome\/toxic epidermal necrolysis combined with haemophagocytic lymphohistiocytosis: a case report","volume":"45","author":"Nic Ionmhain","year":"2017","journal-title":"Anaesth Intensive Care"},{"key":"2021093015370270600_CIT0012","doi-asserted-by":"crossref","first-page":"857","DOI":"10.1016\/j.jdcr.2019.07.022","article-title":"Stevens\u2013Johnson syndrome complicated by fatal hemophagocytic lymphohistiocytosis","volume":"5","author":"Vengalil","year":"2019","journal-title":"JAAD Case Rep"},{"key":"2021093015370270600_CIT0013","doi-asserted-by":"crossref","first-page":"815","DOI":"10.1684\/ejd.2012.1870","article-title":"Toxic epidermal necrolysis complicated by sepsis, haemophagocytic syndrome, and severe liver dysfunction associated with elevated interleukin-10 production","volume":"22","author":"Yamaoka","year":"2012","journal-title":"Eur J Dermatol"},{"key":"2021093015370270600_CIT0014","doi-asserted-by":"crossref","first-page":"1521","DOI":"10.1097\/CCM.0b013e31821201ed","article-title":"Toxic epidermal necrolysis and Stevens\u2013Johnson syndrome: a review","volume":"39","author":"Gerull","year":"2011","journal-title":"Crit Care Med"},{"key":"2021093015370270600_CIT0015","doi-asserted-by":"crossref","first-page":"484","DOI":"10.1016\/j.mayocp.2013.12.012","article-title":"Prognostic factors and outcomes of adults with hemophagocytic lymphohistiocytosis","volume":"89","author":"Parikh","year":"2014","journal-title":"Mayo Clin Proc"},{"key":"2021093015370270600_CIT0016","doi-asserted-by":"crossref","first-page":"4537","DOI":"10.1177\/0300060519868594","article-title":"Stevens\u2013Johnson syndrome and acute vanishing bile duct syndrome after the use of amoxicillin and naproxen in a child","volume":"47","author":"Li","year":"2019","journal-title":"J Int Med Res"},{"key":"2021093015370270600_CIT0017","doi-asserted-by":"crossref","first-page":"e450","DOI":"10.1111\/jdv.14290","article-title":"A case of toxic epidermal necrolysis and vanishing bile duct syndrome, requiring liver transplantation","volume":"31","author":"Momen","year":"2017","journal-title":"J Eur Acad Dermatol Venereol"},{"key":"2021093015370270600_CIT0018","doi-asserted-by":"crossref","first-page":"74","DOI":"10.1016\/j.alit.2015.09.001","article-title":"Retrospective analysis of Stevens\u2013Johnson syndrome and toxic epidermal necrolysis in 87 Japanese patients\u2014treatment and outcome","volume":"65","author":"Yamane","year":"2016","journal-title":"Allergol Int"},{"key":"2021093015370270600_CIT0019","doi-asserted-by":"crossref","first-page":"1340","DOI":"10.1053\/j.gastro.2015.03.006","article-title":"Features and Outcomes of 899 patients with drug-induced liver injury: the DILIN prospective study","volume":"148","author":"Chalasani","year":"2015","journal-title":"Gastroenterology"},{"key":"2021093015370270600_CIT0020","doi-asserted-by":"crossref","first-page":"1062","DOI":"10.4103\/0366-6999.204929","article-title":"Retrospective analysis of Stevens\u2013Johnson syndrome and toxic epidermal necrolysis in 88 Chinese patients","volume":"130","author":"Wang","year":"2017","journal-title":"Chin Med J (Engl)"},{"key":"2021093015370270600_CIT0021","doi-asserted-by":"crossref","first-page":"1634","DOI":"10.1016\/j.burns.2018.12.018","article-title":"Combination therapy: etanercept and intravenous immunoglobulin for the acute treatment of Stevens\u2013Johnson syndrome\/toxic epidermal necrolysis","volume":"45","author":"Pham","year":"2019","journal-title":"Burns"},{"key":"2021093015370270600_CIT0022","doi-asserted-by":"crossref","first-page":"66","DOI":"10.1080\/09546634.2019.1577548","article-title":"Biologic TNF-alpha inhibitors in the treatment of Stevens\u2013Johnson syndrome and toxic epidermal necrolysis: a systemic review","volume":"31","author":"Zhang","year":"2020","journal-title":"J Dermatolog Treat"},{"key":"2021093015370270600_CIT0023","doi-asserted-by":"crossref","first-page":"135","DOI":"10.2147\/JIR.S160964","article-title":"A meta-analysis of cyclosporine treatment for Stevens\u2013Johnson syndrome\/toxic epidermal necrolysis","volume":"11","author":"Ng","year":"2018","journal-title":"J Inflamm Res"},{"key":"2021093015370270600_CIT0024","doi-asserted-by":"crossref","first-page":"177","DOI":"10.1023\/A:1013296310944","article-title":"Efficacy of early immunosuppressive therapy in a child with carbamazepine-associated vanishing bile duct and Stevens\u2013Johnson syndromes","volume":"47","author":"Garcia","year":"2002","journal-title":"Dig Dis Sci"}],"container-title":["Journal of Burn Care &amp; Research"],"original-title":[],"language":"en","link":[{"URL":"http:\/\/academic.oup.com\/jbcr\/advance-article-pdf\/doi\/10.1093\/jbcr\/irab058\/38038051\/irab058.pdf","content-type":"application\/pdf","content-version":"am","intended-application":"syndication"},{"URL":"http:\/\/academic.oup.com\/jbcr\/article-pdf\/42\/5\/1043\/40484175\/irab058.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"syndication"},{"URL":"http:\/\/academic.oup.com\/jbcr\/article-pdf\/42\/5\/1043\/40484175\/irab058.pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2021,9,30]],"date-time":"2021-09-30T18:18:17Z","timestamp":1633025897000},"score":1,"resource":{"primary":{"URL":"https:\/\/academic.oup.com\/jbcr\/article\/42\/5\/1043\/6255376"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2021,4,27]]},"references-count":24,"journal-issue":{"issue":"5","published-online":{"date-parts":[[2021,4,27]]},"published-print":{"date-parts":[[2021,9,30]]}},"URL":"https:\/\/doi.org\/10.1093\/jbcr\/irab058","relation":{},"ISSN":["1559-047X","1559-0488"],"issn-type":[{"value":"1559-047X","type":"print"},{"value":"1559-0488","type":"electronic"}],"subject":[],"published-other":{"date-parts":[[2021,9,1]]},"published":{"date-parts":[[2021,4,27]]}}}