Prolonged Extracorporeal Membrane Oxygenation Support In a Patient with Drug Reaction with Eosinophilia and Systemic Symptoms Syndrome-associated Fulminant Myocarditis - A Case Report and Literature Review.

Abstract

Introduction: Myocarditis is a rare presentation of drug reaction with eosinophilia and systemic symptoms (DRESS) syndrome, traditionally associated with poor clinical outcomes. Prompt recognition and treatment are crucial. Case presentation: A 16-year-old patient presented with acute chest pain, hypotension, and pulmonary oedema 2 months after being diagnosed with trimethoprim/sulfamethoxazole-induced DRESS syndrome. Typical DRESS features were absent at onset of these symptoms. Echocardiography demonstrated biventricular systolic dysfunction, and electrocardiography (ECG) showed complete right bundle branch block and diffuse ST-segment elevation. The patient was admitted for high-dose inotropic support; however, his condition deteriorated. Veno-arterial extracorporeal membrane oxygenation (VA-ECMO) and intra-aortic balloon pump were initiated on the third day of admission. Endomyocardial biopsy suggested the diagnosis of DRESS-associated myocarditis and a high-dose corticosteroid was commenced. While he was ECMO-dependent, the patient suffered multiple episodes of ventricular tachycardia on a background of cardioversion-resistant accelerated idioventricular rhythm. After 24 days of ECMO support, the ventricular function improved, and ECG reverted to sinus rhythm. His recovery allowed corticosteroid discontinuation 15 months after hospital discharge. Conclusion: Typical DRESS syndrome features may not accompany the onset of DRESS-associated myocarditis. Mechanical circulatory support and adequate immunosuppression could save patients with malignant arrhythmias and delayed myocardial recovery.