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1/30. Panclavicular ankylosis in pustulotic arthroosteitis. A case report.

    A 54-year-old man who had palmoplantar pustulosis showed bilateral, complete osseous fusion of the sternoclavicular and acromioclavicular joints. No significant abnormality was seen in the glenohumeral joint. The left clavicle had a nonunion develop, whereas the right did not. The elevation of the right shoulder was limited to 100 degrees in active and passive measurements. The total external rotation and internal rotation at the side was 135 degrees. This decreased to 90 degrees at 60 degrees elevation and to 10 degrees at maximum (100 degrees) elevation. Based on the kinematic data on normal shoulders, it was thought that the ankylosis of both ends of the clavicle held the scapula unrotated during the arm movement so that, at the arm to trunk angle of 100 degrees, the position of the humerus relative to the scapula was equivalent to that of a normal shoulder in complete elevation. The current case provided an extremely rare clinical setting where shoulder mobility depended only on glenohumeral motion as a result of the complete loss of scapulothoracic motion. ( info)

2/30. The sternoclavicular syndrome: experience from a district general hospital and results of a national postal survey.

    OBJECTIVE: To report our local experience of the sternoclavicular syndrome and sample the experience of other rheumatologists in the UK. methods: We studied case records of 23 patients referred to the Southend rheumatology clinic and data obtained from a postal questionnaire survey of British rheumatologists. RESULTS: We describe 58 cases (20 males and 38 females, mean age 47.2 yr). The disease was unilateral in 40 patients. shoulder and/or arm pain (38 cases) with limitation of shoulder movements was an important presenting feature; other presenting features were anterior chest wall pain (14 cases) and neck pain (15 cases). Peripheral joint involvement was seen in 12 cases. skin rash was reported in 12 cases (psoriasis, 6; acne, 2; none had pustulosis). No patients had symptoms or signs of sacroiliitis, and HLA-B27 was negative in 22 out of 23 patients. 99Technetium scintiscanning showed increased uptake in the sternoclavicular region in 31/34 patients (91.1%), but not in the sacroiliac areas. Plain radiographs were abnormal in 18 cases (sclerosis, 9; erosions, 2; soft tissue swelling, 2; bony expansion, 5). CT and/or MRI scans (available in 27 cases) showed erosions in 12 and osteitis in 18. Available histology showed a variable picture, including inflammation, bone erosion, sterile osteomyelitis and fibrosis. The majority of patients (45) were treated with non-steroidal anti-inflammatory drugs: 12 received steroids and 10 received disease-modifying anti-rheumatic drugs (methotrexate, 4; sulphasalazine, 6). Follow-up information was available for 38 patients, of whom 14 became asymptomatic and 24 had chronic disease with intermittent flares. CONCLUSION: Sternoclavicular disease is not uncommon in the UK. It can present with pain in the shoulder, neck or anterior chest wall, and may be underdiagnosed. Our results do not show a link with acne or pustulosis. Features of spondyloarthropathies, such as sacroiliitis and HLA-B27 positivity, were rare in this survey. ( info)

3/30. Sterno-costo-clavicular hyperostosis.

    Sterno-costo-clavicular hyperostosis (SCCH) is a condition of unknown aetiology in which a chronic inflammatory osteitis affects predominantly the medical clavicle, sternum and distal tibia and femur, sometimes associated with skin disease. We report on two patients with the typical bony lesions of SCCH and no associated skin disease. ( info)

4/30. Sternocostoclavicular hyperostosis. Presentation and long-term follow-up of three cases.

    Sternocostoclavicular hyperostosis is a rare disease characterized by recurrent pain and skeletal swelling in the upper part of the chest. The clinical manifestations are closely linked to pustulosis palmo-plantaris but the etiology is still obscure. We present three cases of sternocostoclavicular hyperostosis with a follow-up period of 9-22 years at our department. ( info)

5/30. Stress fracture of the clavicle associated with sternocostoclavicular hyperostosis.

    We report a case of stress fracture of the clavicle associated with sternocostoclavicular hyperostosis. A 60-year-old man sustained a stress fracture of the right clavicle with no history of trauma. On radiography, hyperostosis of the anterior chest wall and ankylosis of the sternoclavicular joint were evident in addition to the fracture. fracture healing was uneventful after 2.5 months. ankylosis of the sternoclavicular joint may have caused increased stress at the midshaft of the clavicle by daily activity or minor trauma. Such a fracture is a rare complication of sternocostoclavicular hyperostosis. ( info)

6/30. Rapid pain relief and remission of sternocostoclavicular hyperostosis after intravenous ibandronate therapy.

    Sternocostoclavicular hyperostosis (SCCH) is an infrequent but painful, localized disturbance of bone metabolism of unknown etiology. The diagnosis of SCCH is generally one of exclusion, and it is therefore frequently missed or delayed, leaving patients with pain that frequently fails to respond to standard analgesic therapy. Consequently, SCCH leads to significantly impaired quality of life. Characteristic increased localized bone turnover and inflammatory osteitis provide a strong rationale for using intravenous bisphosphonates to treat the condition. We report on three patients with long-standing, treatment-refractory SCCH in whom intravenous ibandronate injections (a single administration of 4 mg followed by 2 mg every 3 months for up to a year) produced prompt, dramatic, persistent pain relief and resolution of the other symptoms of the disease. We also review recent evidence suggesting that SCCH is more common than generally believed and that technetium-99 bone scanning can aid in making an accurate diagnosis. ( info)

7/30. 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. ( info)

8/30. calcitonin treatment for intersternocostoclavicular ossification: clinical experience in two cases.

    Intersternocostoclavicular ossification is a benign arthro-osteitis of the upper anterior chest of unknown cause. Two patients with acute exacerbation of this disorder were successfully treated with intramuscular injections of an eel calcitonin analogue (40 units three times a week). Besides symptomatic relief of local pain and swelling, serial scintigrams showed quantitative improvement in radiophosphonate uptake. The rapid alleviation of pain implies that the hormone has a central analgesic effect, in addition to its direct influence on bone cells and antiinflammatory action. In one patient the disease was associated with palmoplantar pustulosis, which was cured with oral colchicine, whereas the other patient did not have such skin lesions. Despite a hypothetical link between palmoplantar pustulosis and intersternocostoclavicular ossification, colchicine had no beneficial impact on the bone pain. salmon calcitonin delivered by nasal spray was tried for the second patient but failed, probably because of insufficient drug delivery. The initial favourable results described here warrant future use of calcitonin injection on a larger number of patients with intersternocostoclavicular ossification. ( info)

9/30. Pustular osteoarthropathy and its differential diagnosis.

    A combination of costo-sterno-clavicular hyperostosis and palmo-plantar pustulosis, sometimes with hyperostotic spondylosis and spondylarthritis, is called pustular osteoarthropathy. In the Western hemisphere 40 cases have been reported and in the far east the condition occurs more commonly. Five cases are described. diagnosis is difficult because the skin lesions can occur before the costo-sterno-clavicular hyperostoses. The clinical condition is discussed together with the differential diagnosis. Pustular osteoarthropathy seems to be an enthesopathy and the palmoplantar pustulosis is interpreted as a form of psoriasis. ( info)

10/30. sternoclavicular joint swellings: diagnosis and management.

    Five patients with sternoclavicular swellings are described. The group presents a variety of diagnoses which highlight the need for thorough investigation and appropriate management of swellings around the sternoclavicular joint. Although frequently assumed to be benign, this series demonstrates the potential occurrence of malignant disease, and the dangers of pursuing a simple conservative course. Conversely, a substantiated benign diagnosis may avoid the use of unnecessary surgical treatment. ( info)
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