SAPHO syndrome with affection of the mandible: diagnosis, treatment, and review of literature

Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2011 Feb;111(2):190-5. doi: 10.1016/j.tripleo.2010.04.037. Epub 2010 Jul 31.

Abstract

The synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome is a rare syndrome of unknown etiology. Involvement of the mandible is found in almost 10% of cases. In the literature, conservative treatment is recommended most often, because decortication and partial resection are found to be ineffective and of temporary profit. We report a case of SAPHO syndrome in a 44-year-old women with unilateral hyperostosis of the mandible and massive painful swelling of the surrounding soft tissues. Owing to facial disfiguration and pain, resection of the affected bone followed by immediate reconstruction with a microvascular iliac crest flap were performed. The aim of this paper was to present the necessity of surgical intervention in SAPHO syndrome of the mandible in cases of esthetic and functional limitation.

Publication types

  • Case Reports
  • Review

MeSH terms

  • Acquired Hyperostosis Syndrome / complications*
  • Acquired Hyperostosis Syndrome / diagnosis
  • Acquired Hyperostosis Syndrome / surgery
  • Adult
  • Bone Transplantation
  • Facial Asymmetry / diagnosis
  • Facial Asymmetry / etiology*
  • Facial Asymmetry / surgery
  • Female
  • Functional Laterality
  • Humans
  • Ilium / transplantation
  • Mandibular Diseases / diagnosis
  • Mandibular Diseases / etiology*
  • Mandibular Diseases / surgery
  • Surgical Flaps
  • Treatment Outcome