PMID- 31020177 OWN - NLM STAT- PubMed-not-MEDLINE LR - 20220408 IS - 2514-2119 (Electronic) IS - 2514-2119 (Linking) VI - 2 IP - 4 DP - 2018 Dec TI - Refractory cardiac myocarditis associated with drug rash with eosinophilia and systemic symptoms syndrome due to anti-bipolar disorder drugs: a case report. PG - yty100 LID - 10.1093/ehjcr/yty100 [doi] LID - yty100 AB - BACKGROUND: Drug rash with eosinophilia and systemic symptoms (DRESS) syndrome is a severe adverse drug reaction accompanied by multiple organ dysfunction. Myocarditis is a manifestation, and once acute necrotizing eosinophilic myocarditis (ANEM) develops, the mortality rate is high. CASE SUMMARY: We report the case of a 37-year-old man who developed myocarditis associated with DRESS syndrome after starting treatment with lithium and quetiapine for bipolar disorder. At that time, he presented with fever, morbilliform eruption, lymphadenopathy, eosinophilia with atypical lymphocytes, and liver dysfunction; bipolar drugs were discontinued and oral prednisolone begun. Four months later, he was admitted to our institution with worsening skin rash and dyspnoea. Transthoracic echocardiography revealed reduced systolic function in both ventricles, and endocardial biopsy indicated hypersensitivity myocarditis. Cardiac function was temporarily normalized by high-dose prednisolone. However, the inflammation was persistent as shown by a re-elevation of troponin T and fall of left ventricular ejection fraction several months later; in addition, (18)F-fluoro-deoxyglucose positron emission tomography with chest computed tomography (FDG-PET/CT) showed focal FDG uptake in the left ventricle. Despite additional treatment with mycophenolate mofetil, the cardiac function deteriorated further, and the patient eventually manifested refractory heart failure classified as New York Heart Association (NYHA) Class III. Myocardial biopsy showed myocyte necrosis associated with ANEM. DISCUSSION: This is the first case report of DRESS-associated myocarditis due to treatment for bipolar disorder. Although the pathophysiology remains incompletely understood, lithium and/or quetiapine can induce refractory myocarditis in DRESS syndrome. Regular measurements of troponin T and FDG-PET/CT are useful for assessing disease progression in DRESS-associated myocarditis. FAU - Hagiwara, Hikaru AU - Hagiwara H AD - Department of Cardiovascular Medicine, Faculty of Medicine and Graduate School of Medicine, Hokkaido University, Kita-15 Nishi-7, Kita-Ku, Sapporo, Japan. FAU - Fukushima, Arata AU - Fukushima A AD - Department of Cardiovascular Medicine, Faculty of Medicine and Graduate School of Medicine, Hokkaido University, Kita-15 Nishi-7, Kita-Ku, Sapporo, Japan. FAU - Iwano, Hiroyuki AU - Iwano H AD - Department of Cardiovascular Medicine, Faculty of Medicine and Graduate School of Medicine, Hokkaido University, Kita-15 Nishi-7, Kita-Ku, Sapporo, Japan. FAU - Anzai, Toshihisa AU - Anzai T AD - Department of Cardiovascular Medicine, Faculty of Medicine and Graduate School of Medicine, Hokkaido University, Kita-15 Nishi-7, Kita-Ku, Sapporo, Japan. LA - eng PT - Case Reports DEP - 20181010 PL - England TA - Eur Heart J Case Rep JT - European heart journal. Case reports JID - 101730741 PMC - PMC6426116 OTO - NOTNLM OT - Acute necrotizing eosinophilic myocarditis (ANEM) OT - Bipolar disorder OT - Case report OT - Drug rash with eosinophilia and systemic symptoms (DRESS) syndrome OT - Heart failure OT - Lithium OT - Quetiapine EDAT- 2019/04/26 06:00 MHDA- 2019/04/26 06:01 PMCR- 2018/10/10 CRDT- 2019/04/26 06:00 PHST- 2018/03/24 00:00 [received] PHST- 2018/08/25 00:00 [accepted] PHST- 2019/04/26 06:00 [entrez] PHST- 2019/04/26 06:00 [pubmed] PHST- 2019/04/26 06:01 [medline] PHST- 2018/10/10 00:00 [pmc-release] AID - yty100 [pii] AID - 10.1093/ehjcr/yty100 [doi] PST - epublish SO - Eur Heart J Case Rep. 2018 Oct 10;2(4):yty100. doi: 10.1093/ehjcr/yty100. eCollection 2018 Dec.