Cryopyrin-associated periodic syndrome

Pediatr Dermatol. 2014 Mar-Apr;31(2):228-31. doi: 10.1111/j.1525-1470.2012.01812.x. Epub 2012 Aug 14.

Abstract

Cryopyrin-associated periodic syndromes (CAPS) are characterized by apparently unprovoked attacks of fever, rashes, and musculoskeletal and sensorineural inflammation accompanied by high acute-phase reactants. Excessive interleukin-1 (IL-1) signaling appears to be a constant feature in the pathomechanism of the disease, driven by a gain-of-function mutation in the NLRP3 gene. Herein, we present the case of a 9-month-old boy with recurrent nonpruritic rashes and episodes of fever. The difficulties of early diagnosis due to initially mild clinical symptoms and the dramatic response to anti-IL-1 therapy after diagnosis emphasize the practical relevance of considering CAPS as a differential diagnosis in these patients.

Publication types

  • Case Reports

MeSH terms

  • Antirheumatic Agents / therapeutic use
  • Cryopyrin-Associated Periodic Syndromes / diagnosis*
  • Cryopyrin-Associated Periodic Syndromes / drug therapy
  • Cryopyrin-Associated Periodic Syndromes / genetics
  • Diagnosis, Differential
  • Humans
  • Infant
  • Interleukin 1 Receptor Antagonist Protein / therapeutic use
  • Male

Substances

  • Antirheumatic Agents
  • Interleukin 1 Receptor Antagonist Protein