Follow-up and quality of life of patients with cryopyrin-associated periodic syndromes treated with Anakinra

J Pediatr. 2010 Aug;157(2):310-315.e1. doi: 10.1016/j.jpeds.2010.02.040. Epub 2010 May 15.

Abstract

Objective: To evaluate the quality of life and long-term follow-up of patients enrolled in the Italian registry of cryopyrin-associated periodic syndromes (CAPS).

Study design: Since 2004, 20 patients with CAPS were enrolled in a common registry from different Italian Centers of Pediatric Rheumatology; 14 patients were treated with Anakinra in an open fashion. Both treated and untreated patients were routinely followed according to standard of care. The Child Health Questionnaire (CHQ-PF 50) was used to assess the health-related quality of life.

Results: The mean duration of follow-up was 37.5 months. In all treated patients, a complete and persistent control of the inflammatory manifestations was observed with no further progression of the disease. At enrollment in the registry, patients showed a poorer health-related quality of life than healthy children in both physical and the psychosocial summary scores. Treatment was associated with a dramatic and sustained amelioration of a variety of measures of poor quality of life, particularly in those concerning the global health perception, bodily pain-discomfort, and other physical domains.

Conclusions: Long-term IL-1 blockade produces a significant and persistent improvement in the clinical manifestations associated with the disease and on the overall quality of life.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adolescent
  • Adult
  • Case-Control Studies
  • Child
  • Child, Preschool
  • Cryopyrin-Associated Periodic Syndromes / drug therapy*
  • Cryopyrin-Associated Periodic Syndromes / physiopathology*
  • Female
  • Follow-Up Studies
  • Humans
  • Inflammation
  • Interleukin 1 Receptor Antagonist Protein / therapeutic use*
  • Interleukin-1 / antagonists & inhibitors
  • Male
  • Phenotype
  • Quality of Health Care
  • Quality of Life
  • Surveys and Questionnaires
  • Syndrome
  • Treatment Outcome

Substances

  • Interleukin 1 Receptor Antagonist Protein
  • Interleukin-1