Fatal sulfasalazine-induced eosinophilic myocarditis in a patient with periodic fever syndrome

Med Princ Pract. 2015;24(2):195-7. doi: 10.1159/000369584. Epub 2014 Dec 16.

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-year-old 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.

Publication types

  • Case Reports
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adult
  • Anti-Inflammatory Agents, Non-Steroidal / adverse effects*
  • Anti-Inflammatory Agents, Non-Steroidal / therapeutic use
  • Arthritis / drug therapy
  • Autopsy
  • Eosinophilia / chemically induced
  • Fatal Outcome
  • Fever / complications
  • Humans
  • Male
  • Myocarditis / chemically induced*
  • Myocarditis / complications
  • Myocarditis / pathology
  • Shock, Cardiogenic / complications
  • Sulfasalazine / adverse effects*
  • Sulfasalazine / therapeutic use

Substances

  • Anti-Inflammatory Agents, Non-Steroidal
  • Sulfasalazine