Author/Authors :
Jeremic, Ivica University of Belgrade - Faculty of Medicine, Institute of Rheumatology, Clinical Center of Serbia, Serbia , Vujasinovic-Stupar, Nada University of Belgrade - Faculty of Medicine, Institute of Rheumatology, Clinical Center of Serbia, Serbia , Terzic, Tatjana University of Belgrade - Faculty of Medicine, Institute of Pathology, Clinical Center of Serbia, Serbia , Damjanov, Nemanja University of Belgrade - Faculty of Medicine, Institute of Rheumatology, Clinical Center of Serbia, Serbia , Nikolic, Milos University of Belgrade - Faculty of Medicine, Clinical Center of Serbia, Clinic of Dermatovenereology, Serbia , Bonaci-Nikolic, Branka University of Belgrade - Faculty of Medicine, Clinical Center of Serbia, Clinic of Allergy and Clinical Immunology, Serbia
Abstract :
Objective: The aim of this paper is to report the first case of drug-induced eosinophilic myocarditis (EM) in a patient with hereditary periodic fever syndrome (PFS). Case: A 28-yearold man with hyper-IgD syndrome, one of the PFS, developed a sulfasalazine-induced systemic hypersensitivity reaction complicated by EM. Thirteen days after sulfasalazine introduction, which had been given for arthritis, the patient developed fever, facial/neck edema, rash and cardiogenic shock, and died within 8 h. The autopsy revealed hemophagocytosis, while acute heart failure caused by necrotizing EM was established as the cause of death. Conclusion: This was a case of drug-induced EM in a patient with PFS that had an atypical presentation, rapid evolution and poor outcome.
Keywords :
Eosinophilic myocarditis · Drug , induced hypersensitivity syndrome · Sulfasalazine · Hyper , IgD syndrome · Periodic fever syndrome