Cases reported "Rheumatic Diseases"

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11/63. Serious infections associated with anticytokine therapies in the rheumatic diseases.

    The ability to target and neutralize macrophage-derived inflammatory cytokines, particularly tumor necrosis factor-alpha (TNF-alpha), has emerged in recent years as one of the most important advances in the treatment of rheumatoid arthritis, Crohn's disease, and several other systemic inflammatory diseases. In rheumatoid arthritis, for example, these biological agents rapidly reduce signs and symptoms of joint inflammation and profoundly slow the progression of joint damage. However, data that have emerged following food and Drug Administration approval of these agents have alerted clinicians to an increased likelihood of opportunistic infections in patients treated with these agents, particularly tuberculosis. The effect of TNF inhibition on the frequency of infection with more common bacterial pathogens is less clear. Animal models of tuberculosis and other opportunistic infections have demonstrated the importance of TNF-alpha in controlling and containing intracellular pathogens. The spectrum of infections reported to date in the setting of anti-TNF-alpha treatment is reviewed here. In addition, relevant animal data illustrating potential mechanistic roles for TNF-alpha in host responses to infection are also reviewed.
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12/63. Prevention of amputation caused by rheumatic diseases following a novel therapy of exposing bone marrow, occlusive dressing and subsequent epidermal grafting.

    BACKGROUND: Wounds with exposed bones caused by rheumatic diseases commonly result in amputation despite progress in our understanding of wound-healing mechanisms. OBJECTIVES: To determine whether an experimental therapy of bone marrow exposure, an occlusive dressing and subsequent grafting of epidermal sheets accelerates healing and reduces the need for amputation in patients with rheumatic diseases. methods: Fifteen patients, including those with rheumatoid arthritis or systemic sclerosis, who had wounds with exposed bones were treated either with the standard procedure, consisting of local wound care, debridement with a scalpel, bed rest and parenteral antibiotics (n = 8), or with a newly developed experimental procedure (n = 7). In that new procedure, the affected bone was initially exposed by debridement with a scalpel, followed by partial excision with a bone scraper until bleeding was observed from the exposed bone. The lesions were immediately covered with an occlusive dressing, and were eventually treated with epidermal grafts obtained from suction blisters. RESULTS: A comparison with standard therapy demonstrated that the time needed for wound healing was similar, but that the newly developed combination therapy reduced the risk of amputation (P = 0.020). No skin ulcers or erosions were observed for at least 1 year in five of seven patients (72%) due to the adoption of stable palmoplantar-type characteristics in grafts derived from the trunk epidermis. CONCLUSIONS: Our study indicates that exposure of bone marrow cells plus an occlusive dressing accelerates the healing of skin ulcers at least partly through the preparation of a healthy well-granulated wound bed and that subsequent epidermal grafting achieves site-specific differentiation through epithelial-mesenchymal interactions.
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13/63. Massive calcaneal enthesopathy in a non-healing leg ulcer: a case report.

    Enthesopathy at the superior or inferior surface of a calcaneus may be seen in normal individuals having degenerative osteoarthrosis. This condition is also known to occur in patients with rheumatoid arthritis, seronegative spondyloarthropathy, trauma, as well as inflammatory and metabolic diseases. Enthesopathy may sometimes be the first manifestation of a variety of rheumatic diseases. In this report, we present a case of massive enthesopathy of the superior and inferior surface of the calcaneus giving rise to an 'axe effect'.
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14/63. Influence of pregnancy on disease activity in a patient with palindromic rheumatism.

    A 30-year-old woman with a 12-year history of palindromic rheumatism without progression to chronic arthritis experienced pronounced reduction of arthritic symptoms during both of her 2 pregnancies with, on each occasion, symptoms recurring 3 months after delivery. This is the first reported case suggesting an ameliorating effect of pregnancy on disease activity in palindromic rheumatism.
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15/63. rheumatic diseases and chronic myelogenous leukemia, presentation of four cases and review of the literature.

    We report four patients with rheumatic disease (RD) and chronic myelogenous leukemia (CML). In two patients with Behcet's disease (BD) and rheumatoid arthritis (RA), CML developed after RD, in two patients with diffuse cutaneous systemic sclerosis and spondyloarthropathy, RD was diagnosed after CML. A variety of interactions have been described between hematological malignancies and RD. Nevertheless, few cases of RD have been documented associated with CML. It is unclear whether the development of CML in patients with RD and RD development after CML occurs by chance alone, is due to the underlying disease, or is facilitated by drugs. Whatever the cause is, it should be kept in mind that CML may develop in the course of RD and RD may be seen in CML patients.
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16/63. Fibroblastic rheumatism.

    We report the first known South African case of fibroblastic rheumatism, a rare dermatoarthropathy. Our patient presented with the typical clinical features of a sudden-onset, erosive polyarthritis with cutaneous nodules and sclerodactyly. Significant functional loss occurred within a period of 3 to 4 months. x-rays of the hands showed a single erosion, whereas magnetic resonance imaging showed further erosions as well as soft tissue and synovial enhancement. The unique histologic findings of fibroblastic proliferation, thickened collagen, and dermal fibrosis confirmed the diagnosis of fibroblastic rheumatism. Our patient was treated with a combination of methotrexate and oral prednisolone with subsequent resolution of her synovitis/arthritis and no further progression of her sclerodactyly and associated functional loss.The course of fibroblastic rheumatism is known to vary and although multiple therapeutic options have been tried, the question remains whether any of the therapies alters the natural course of the disease. However, considering the probable role of lymphocytes and fibrogenic cytokines, an increased awareness with early diagnosis and treatment in the initial inflammatory stage may prevent the development of incapacitating joint sequelae.
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17/63. Acute aortitis and aortic incompetence due to systemic rheumatological disorders.

    We report the clinical, laboratory and echocardiographic features of five cases of aortic incompetence associated with ankylosing spondylitis, rheumatoid arthritis and undefined connective tissue diseases. immunosuppression with steroids and cytotoxic agents was used to suppress aortic root inflammation in four cases; in three the aortic root size stabilized and the patients remain well with no evidence of increasing aortic incompetence. In one case, control of the inflammatory process was never fully achieved for any length of time and the patient died shortly after aortic valve replacement. A fifth case required urgent valve replacement and remains well. A systemic rheumatological disorder should be considered in cases of apparent "lone" aortic incompetence and conversely aortic incompetence should not be overlooked in established systemic rheumatological disease. Immunosuppressive therapy may prevent or delay the need for aortic valve replacement in such cases.
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18/63. Coexistent Felty's syndrome and palindromic rheumatism.

    Palindromic rheumatism is a syndrome of intermittent abrupt onset monoarthritis with asymptomatic intercritical periods of variable duration, which commonly evolves into rheumatoid arthritis. Felty's syndrome consists of leucopenia (selective neutropenia) and splenomegaly, usually occurring in longstanding classic rheumatoid arthritis. Felty's syndrome can be confused with the more recently recognised rheumatoid arthritis associated large granular lymphocyte proliferative disease. This paper describes a patient with palindromic rheumatism presenting with Felty's syndrome in whom large granular lymphocyte proliferative disease was ruled out by lymphocyte phenotyping.
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19/63. Nodular regenerative hyperplasia of the liver in rheumatic diseases: report of seven cases and review of the literature.

    Nodular regenerative hyperplasia (NRH) of the liver is an uncommon pathologic finding associated, in most cases, with rheumatic and hematologic diseases. Although its pathogenesis remains unclear, NRH probably results from liver regeneration to maintain its functional capacity after ischemia-induced injury. An intrahepatic microvascular occlusive mechanism has been considered most likely pathogenetically. NRH may lead to portal hypertension. Thus, the diagnosis of Felty's syndrome must be considered with caution in patients with rheumatoid arthritis (RA) and NRH of the liver. We report seven additional cases of NRH in patients with rheumatic disorders and review the literature to determine the patterns of clinical presentation and natural history of this condition. We also report four patients (three systemic lupus erythematosus [SLE] and one primary antiphospholipid syndrome [PAPS]) in whom antiphospholipid antibodies may have played a role in the genesis of NRH.
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20/63. Necrotizing vasculitis of the gallbladder and the appendix. Similarity in the morphology of rheumatoid arteritis and polyarteritis nodosa.

    Two patients with rheumatoid arthritis (RA) and polyarteritis nodosa (PN) presented with abdominal pain. Both revealed necrotizing vasculitis of the gallbladder and the appendix respectively. The controversial role of corticosteroids in the pathogenesis of rheumatoid arteritis, the rarity of rheumatoid arteritis of the gallbladder and the morphologic similarity of the vascular lesion in the gallbladder and appendix are discussed.
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