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1/12. thrombosis of the subclavian vein in SAPHO syndrome. A case-report.

    The hallmark of SAPHO Syndrome (synovitis, Acne, Pustulosis, hyperostosis, osteitis) is osteitis of the anterior chest wall, which can be so pronounced as to cause thoracic outlet syndrome, thrombosis of the subclavian vein, and compression of the superior vena cava. Suggestive skin manifestations, namely palmoplantar pustulosis and severe acne, generally antedate the bone and joint lesions. We report a case of SAPHO syndrome that went unrecognized for many years and eventually caused compression and thrombosis of the right subclavian vein requiring cleidectomy. SAPHO syndrome should be considered in every patients with anterior chest wall inflammation, particularly when skin lesions are also present.
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2/12. 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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3/12. SAPHO syndrome or psoriatic arthritis? A familial case study.

    OBJECTIVE: To discuss the relationships between SAPHO (synovitis, acne, pustulosis, hyperostosis and osteitis) syndrome and the group of spondylarthropathies. methods: Few reports of familial SAPHO have been published. We describe three children, two sisters and one brother, whose clinical and radiological presentation was in accordance with SAPHO syndrome. RESULTS: Two children developed psoriasis, and one child palmoplantar pustulosis. Both sacroiliac and sternoclavicular joints were involved in these three cases. Some features in our observations are also common to psoriatic arthritis. No association was found with hla antigens, but a history of trauma preceding the onset of symptoms was present in all three children. CONCLUSIONS: We can consider that SAPHO is nosologically related to spondylarthropathies. Psoriatic arthritis could be the missing link between SAPHO and spondylarthropathies. It is likely that both genetic and environmental factors are involved.
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4/12. SAPHO syndrome: report of three cases and review of the literature.

    SAPHO (synovitis, acne, pustulosis, hyperostosis, and osteitis) is an acronym that describes a seronegative syndrome of bone and joint lesions, frequently localized to the anterior chest wall, associated with variable dermatologic manifestations. Dermatologists should be aware of this entity, as early diagnosis can prevent unnecessary surgery and avoid prolonged ineffective antibiotic therapy. We report three new cases and review the literature.
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5/12. 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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6/12. Palmoplantar pustulosis with pustulotic arthroosteitis involving temporal osteomyelitis and meningitis.

    A 46-year-old man who had been suffering from palmoplantar pustulosis (PPP) for 3 years had anterior chest pain and left temporal pain from six months after the onset of his disease. A bone scan revealed abnormal uptake at the sternoclavicular joint and left temporal region. The head CT and MRI gave the diagnosis of temporal osteomyelitis with meningitis and myositis. His headache continued even after tonsillectomy and was effectively treated with cyclosporine A (3 mg/kg/day). Oral cyclosporine A was beneficial for the osteomyelitis and skin lesions. Sterile lytic bone lesions occurring most often at the sternocostoclavicular joint have been associated with PPP. However, there have been no reports of a PPP patient with temporal osteomyelytic involvement.
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7/12. 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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8/12. The "SAPHO" syndrome: a case report of a patient with unusual bone scan findings.

    PURPOSE: The authors describe the clinical and bone scintigraphic findings of the SAPHO syndrome, which is characterized by synovitis, acne, palmoplantar pustulosis, hyperostosis, and osteitis. The case report illustrates the significance of bone scintigraphy in the diagnosis. It shows that Tc-99m MDP scanning can detect signs of arthritis not seen with other imaging methods, because the arthritis is inflammatory in nature and does not always cause bone erosion. Thus it is not visualized on plain radiographs. knowledge of this disorder may help in the differential diagnosis of arthritis. MATERIALS AND methods: Tc-99m MDP bone scintigraphy was used to diagnose arthritic changes. Whole-body and multiple delayed spot images were obtained in a 39-year-old diabetic, hypertensive woman who had tenderness in the plantar aspect of her heels. Swelling of the small and large joints of the feet, ankles, knees, hips, right sacroiliac joints, and shoulders was noted. She also had hydradenitis suppurativa and a history of a previous episode in which the arthritis improved after surgical treatment for the hydradenitis. RESULTS: All active joint lesions were visualized on the bone scan, including the arthritis, which was not detected with other imaging methods. They were all well demonstrated in the bone scintiscan. The scan findings, along with the presence of hydradenitis, led to the correct diagnosis of SAPHO syndrome. CONCLUSION: Tc-99m MDP bone scanning may be helpful in diagnosing arthritis as associated with the SAPHO syndrome.
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9/12. Bone scintigraphy using Tc-99m DPD and F18-FDG in a patient with SAPHO syndrome.

    synovitis (inflammatory arthritis), acne (pustulosa), palmoplantar pustulosis, hyperostosis and osteitis (bland osteomyelitis) are symptoms forming the acronym SAPHO. We present the case of a 48-year old man with sterno-costo-clavicular hyperostosis and typical pustulosis palmaris. We performed Tc-99m DPD serial bone scanning to monitor the course of disease and to assess therapeutic efficiency. Control bone scans in 2001 showed minor compromises of the ribs and diminished disease activity on collarbones and the sternum after medication with non-steroidal anti-inflammatory drugs (NSAIDs) and bisphosphonates. F-18 FDG PET presented synovial inflammation in the left sterno-clavicular joint but no relevant tracer uptake on clavicles or breastbones. In case of diagnostic doubts F-18 FDG PET could be recommended in order to discriminate bland osteomyelitis from bacterial osteomyelitis or from bone malignancy when SAPHO-syndrome is assumed.
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10/12. In SAPHO syndrome anti-TNF-alpha therapy may induce persistent amelioration of osteoarticular complaints, but may exacerbate cutaneous manifestations.

    OBJECTIVES: SAPHO syndrome (synovitis, acne, pustulosis, hyperostosis and osteitis) is a rare disease combining skin, bone and joint manifestations. In recent years new therapeutic strategies have been tried, among them TNF-alpha-blocking agents. We report our experience with infliximab in four cases of SAPHO syndrome refractory to conventional therapies. methods: Between 2002 and 2005, four cases of SAPHO syndrome (two females and two males; mean age 49.7 yr) responding poorly to conventional drugs were treated with infliximab. The dose was 5 mg/kg, according to the protocol used in spondyloarthropathies, with infusions at 0, 2 and 6 weeks followed by 6 weeks intervals. No active cutaneous manifestations were present at the time of starting therapy. RESULTS: Complete remission of osteoarticular involvement was achieved after the second or third infusion, and the positive response was maintained for up to 12 months. A patient relapsed after discontinuation of infliximab, because of infectious complication. Palmoplantaris pustulosis relapsed in two patients after three and six infusions, respectively; there was slight improvement after discontinuation of anti-TNF-alpha drugs. CONCLUSIONS: Infliximab seems to be a very effective therapy for osteoarticular complaints of SAPHO syndrome. Cutaneous involvement responded less favourably, palmoplantaris pustulosis relapse being a possible complication.
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