PFAPA syndrome is not a sporadic disease

Rheumatology (Oxford). 2010 Oct;49(10):1984-7. doi: 10.1093/rheumatology/keq187. Epub 2010 Jul 2.

Abstract

Objectives: To determine whether PFAPA (periodic fever, aphthous stomatitis, pharyngitis and cervical adenitis) patients have a positive family history (FH) for recurrent fever syndromes.

Method: For all patients with PFAPA seen in two paediatric rheumatology centres (Romandy, Switzerland and Bordeaux, France), parents were interviewed to record the FH for periodic fever. As controls, we interviewed a group of children without history of recurrent fever.

Results: We recruited 84 patients with PFAPA and 47 healthy children. The FH for recurrent fever (without an infectious cause and recurring for at least half a year) was positive in 38/84 (45%), and was positive for PFAPA (diagnosis confirmed by a physician) in 10/84 (12%) of the PFAPA patients. For 29 of the 38 patients with positive FH, the affected person was a sibling or a parent. None of the healthy children had a positive FH for recurrent fever or PFAPA. A positive FH for rheumatological diseases was seen in both groups of children.

Conclusion: These data show that a significant percentage of PFAPA patients present a positive FH of recurrent fever and PFAPA. This familial susceptibility suggests a potential genetic origin for this syndrome.

MeSH terms

  • Adolescent
  • Adult
  • Case-Control Studies
  • Child
  • Child, Preschool
  • Diagnosis, Differential
  • Female
  • Fever of Unknown Origin / diagnosis*
  • Fever of Unknown Origin / etiology
  • Humans
  • Lymphadenitis / diagnosis*
  • Lymphadenitis / etiology
  • Male
  • Periodicity
  • Pharyngitis / diagnosis*
  • Pharyngitis / etiology
  • Recurrence
  • Stomatitis, Aphthous / diagnosis*
  • Stomatitis, Aphthous / etiology
  • Syndrome
  • Young Adult