Severe destruction of the temporomandibular joint with complete resorption of the condyle associated with synovitis, acne, pustulosis, hyperostosis, and osteitis syndrome

Oral Surg Oral Med Oral Pathol Oral Radiol. 2013 Aug;116(2):e128-33. doi: 10.1016/j.oooo.2013.03.011. Epub 2013 May 8.

Abstract

The synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome consists of a combination of inflammatory bone disorders and dermatologic pathology. Bone lesions as a form of diffuse sclerosing osteomyelitis in the mandible occur in the posterior body and ramus. Bone lesions rarely spread to the temporomandibular joint (TMJ) where ankylosis may result. Herein we present an unusual case of SAPHO syndrome with TMJ involvement in which severe destruction of the TMJ occurred. We observed an extension of the invasive soft tissue lesion into the infratemporal fossa from the TMJ with complete resorption of the condyle. In contrast to other previously reported cases, in our case the condyle was strongly suspected as the primary site of the bone lesion with subsequent extension to the ramus and infratemporal fossa. The destructive nature and related symptoms resembled a malignant tumor.

Publication types

  • Case Reports
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Acquired Hyperostosis Syndrome / complications*
  • Acquired Hyperostosis Syndrome / diagnosis
  • Anti-Inflammatory Agents / therapeutic use
  • Bone Resorption / etiology*
  • Diagnosis, Differential
  • Follow-Up Studies
  • Glucocorticoids / therapeutic use
  • Humans
  • Imaging, Three-Dimensional / methods
  • Male
  • Mandibular Condyle / pathology*
  • Mandibular Diseases / etiology*
  • Middle Aged
  • Osteomyelitis / etiology
  • Osteosclerosis / etiology
  • Prednisolone / therapeutic use
  • Radiography, Panoramic
  • Temporomandibular Joint Disorders / etiology*
  • Tomography, X-Ray Computed / methods

Substances

  • Anti-Inflammatory Agents
  • Glucocorticoids
  • Prednisolone