title: Severe destruction of the temporomandibular joint with complete resorption of the condyle associated with synovitis, acne, pustulosis, hyperostosis, and osteitis syndrome.
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.
authors.affiliation: Division of Oral and Maxillofacial Surgery, Niigata University Graduate School of Medical and Dental Sciences, Niigata, Japan. damadama@dent.niigata-u.ac.jp
authors.forename: Yasumitsu
authors.initials: Y
authors.lastname: Kodama
authors.forename: Ray
authors.initials: R
authors.lastname: Tanaka
authors.forename: Akira
authors.initials: A
authors.lastname: Kurokawa
authors.forename: Hisashi
authors.initials: H
authors.lastname: Ohnuki
authors.forename: Sara
authors.initials: S
authors.lastname: Sultana
authors.forename: Takafumi
authors.initials: T
authors.lastname: Hayashi
authors.forename: Tateyuki
authors.initials: T
authors.lastname: Iizuka
authors.forename: Ritsuo
authors.initials: R
authors.lastname: Takagi
mesh_terms: D020083:Acquired Hyperostosis Syndrome / Q000150:complications* / Q000175:diagnosis; D000893:Anti-Inflammatory Agents / Q000627:therapeutic use; D001862:Bone Resorption / Q000209:etiology*; D003937:Diagnosis, Differential; D005500:Follow-Up Studies; D005938:Glucocorticoids / Q000627:therapeutic use; D006801:Humans; D021621:Imaging, Three-Dimensional / Q000379:methods; D008297:Male; D008335:Mandibular Condyle / Q000473:pathology*; D008336:Mandibular Diseases / Q000209:etiology*; D008875:Middle Aged; D010019:Osteomyelitis / Q000209:etiology; D010026:Osteosclerosis / Q000209:etiology; D011239:Prednisolone / Q000627:therapeutic use; D011862:Radiography, Panoramic; D013705:Temporomandibular Joint Disorders / Q000209:etiology*; D014057:Tomography, X-Ray Computed / Q000379:methods
chemical_list: D000893:Anti-Inflammatory Agents; D005938:Glucocorticoids; D011239:Prednisolone