Paediatric case series of drug reaction with eosinophilia and systemic symptoms (DRESS): 12-year experience at a single referral centre in Hong Kong and the first reported use of infliximab

Eur Ann Allergy Clin Immunol. 2018 Nov;50(6):273-276. doi: 10.23822/EurAnnACI.1764-1489.47. Epub 2017 Nov 30.

Abstract

DRESS (drug reaction with eosinophilia and systemic symptoms) is a rare but potentially life-threatening disorder characterized by fever, skin eruption, haematological abnormalities and multi-organ dysfunction after drug exposure. The pathophysiology is thought to be related to interactions between culprit drugs, viral reactivation and T-lymphocytes activation. We report 4 paediatric patients with DRESS who were treated at our centre over the past 12 years. Most cases improved after corticosteroids. Other immunosuppressive medications were attempted in refractory cases with varied outcomes. Patient 3 was the first reported case that involved the use of infliximab, a TNF-α inhibitor, for DRESS. Although clinical efficacy was not observed for this one patient, a previous study demonstrated that patients with DRESS, disease progression and HHV-6 reactivation had elevated pre-treatment TNF- α and IL-6 levels. Further research is needed to explore the role of these cytokines in DRESS.

Keywords: Chinese; DRESS; Hong Kong; drug reaction with eosinophilia and systemic symptoms; drug-induced hypersensitivity syndrome; paediatric.

Publication types

  • Case Reports

MeSH terms

  • Adolescent
  • Adrenal Cortex Hormones / therapeutic use
  • Child, Preschool
  • Dermatologic Agents / toxicity*
  • Drug Hypersensitivity Syndrome / diagnosis
  • Drug Hypersensitivity Syndrome / drug therapy*
  • Drug Hypersensitivity Syndrome / epidemiology*
  • Eosinophilia / chemically induced*
  • Female
  • Hong Kong / epidemiology
  • Humans
  • Infliximab / toxicity*
  • Male
  • Skin Tests
  • Tumor Necrosis Factor-alpha / antagonists & inhibitors

Substances

  • Adrenal Cortex Hormones
  • Dermatologic Agents
  • Tumor Necrosis Factor-alpha
  • Infliximab