Synovitis, acne, pustulosis, hyperostosis, osteitis (SAPHO) syndrome in childhood: a report of ten cases and review of the literature

Eur J Pediatr. 2000 Aug;159(8):594-601. doi: 10.1007/s004310000500.

Abstract

Chronic recurrent multifocal osteomyelitis is a rare chronic inflammatory musculoskeletal process observed in children and young adults. Recently, the acronym SAPHO syndrome (for synovitis, acne, pustulosis, hyperostosis, osteitis) was coined to emphasise the association between osteo-articular inflammations and different skin abnormalities which are aseptic and filled with neutrophils. In adults, chronic recurrent multifocal osteomyelitis is now a classical manifestation of SAPHO syndrome. Chronic skin disorders were seen in eight of ten children on follow-up at the University Children's Hospitals in Bern and Zurich and in 61 of 260 paediatric cases reported in the literature. The different skin lesions were palmoplantar pustulosis (n = 40), non-palmoplantar pustulosis (n = 6), psoriasis vulgaris (n = 16) or severe acne (n = 4). More rarely Sweet syndrome (n = 2) or pyoderma gangrenosum (n = 1) were reported.

Conclusion: The synovitis, acne, pustulosis, hyperostosis, osteitis syndrome is pertinent even in paediatrics since skin involvement is frequent.

Publication types

  • Case Reports
  • Review

MeSH terms

  • Acquired Hyperostosis Syndrome / complications
  • Acquired Hyperostosis Syndrome / diagnosis*
  • Acquired Hyperostosis Syndrome / epidemiology*
  • Adolescent
  • Age Distribution
  • Age of Onset
  • Biopsy
  • Child
  • Child, Preschool
  • Diagnosis, Differential
  • Female
  • Follow-Up Studies
  • Humans
  • Male
  • Pain / etiology
  • Sex Distribution