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1/5. SAPHO syndrome of the temporomandibular joint associated with sudden deafness.

    We report a case of arthritis of the temporomandibular joint (TMJ) associated with sclerosing osteomyelitis of the mandible and temporal bone, causing deafness. The presence of a palmoplantar pustulosis established the diagnosis of SAPHO syndrome. SAPHO (an acronym referring to synovitis, acne, palmoplantar pustulosis, hyperostosis, and osteitis) syndrome is defined by the association of characteristic osteoarticular and dermatologic manifestations, with diffuse sclerosing osteomyelitis of the mandible being a part of this entity. We review the literature of SAPHO syndrome with mandibular manifestations and discuss the mechanisms of inflammatory spread from the TMJ to the cochlea. To our knowledge, this is the first description of skull base involvement in a patient with SAPHO syndrome leading to sudden deafness.
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ranking = 1
keywords = mandible
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2/5. Primary chronic osteomyelitis associated with synovitis, acne, pustulosis, hyperostosis and osteitis (SAPHO syndrome).

    Diffuse sclerosing osteomyelitis may indicate the mandibular localisation of the SAPHO syndrome. Twelve patients with diffuse sclerosis of the mandible were examined for symptoms of the SAPHO syndrome. Nine patients were found to have primary chronic osteomyelitis and eight of these represented a SAPHO syndrome. Results in this series support the hypothesis of an association between primary chronic osteomyelitis and the SAPHO syndrome.
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ranking = 0.5
keywords = mandible
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3/5. Diffuse chronic sclerosing osteomyelitis and the synovitis, acne, pustolosis, hyperostosis, osteitis (SAPHO) syndrome in two sisters.

    Two sisters with diffuse chronic sclerosing osteomyelitis of the mandible and the humerus and the synovitis, acne, pustolosis, hyperostosis and osteitis syndrome (SAPHO syndrome) are presented. The diagnoses of diffuse chronic sclerosing osteomyelitis at the age of 12 years and 27 years, respectively, were based on typical medical history, clinical symptoms and radiographic, histologic and scintigraphic findings. Because skin lesions and scintigraphic enhancement of the sternoclavicular joints with hyperostosis were present, a SAPHO syndrome was diagnosed in both sisters. Microbiological cultures of biopsy specimens revealed coagulase-negative staphylococcus aureus at the humerus and haemophilus parainfluenzae, streptococcus, actinomyces and Veilonella species at the mandible. Repeated operative procedures, including decortications, resection and reconstruction, and multiple histologic and microbiologic studies were performed over a period of up to 20 years. Since HLA typing yielded identical gene loci, we suggest that hereditary and autoimmune factors may play a role in the pathogenesis of these cases.
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ranking = 1
keywords = mandible
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4/5. Acne with chronic recurrent multifocal osteomyelitis involving the mandible as part of the SAPHO syndrome: case report.

    For 12 years, a 26-year-old man had acne conglobata and a non-suppurative diffuse sclerosing osteomyelitis of the mandible as part of a chronic recurrent multifocal osteomyelitis of the sternum, the pelvic bones, and the femoral head, and aseptic arthritis of the knee, the fibulotalar, and the sternoclavicular joints. This fulfills the formal criteria of the SAPHO syndrome. Repeated surgical and antibiotic treatment combined with hyperbaric oxygen caused partial improvement. Complete relief and partial disappearance of the scintigraphic lesions was achieved with long-term corticosteroids, non-steroidal anti-inflammatory drugs, minocycline, and isotretinoin.
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ranking = 2.5
keywords = mandible
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5/5. Scintigraphic evaluation of chronic osteomyelitis of the mandible in SAPHO syndrome.

    A patient with SAPHO syndrome (synovitis, acne, pustulosis, hyperostosis and osteitis) was evaluated by combined scintigraphy. (99m)Tc HMDP scintigraphy showed accumulation in the sternum and lumbar vertebrae as well as the right mandible, whereas (67)Ga citrate showed an accumulation in the right mandible, but not in the sternum or lumbar vertebrae. These results are consistent with chronic osteomyelitis in the mandible.
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ranking = 3.5
keywords = mandible
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