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1/129. Lupus anticoagulant in myelofibrosis.

    A patient with myelofibrosis who also demonstrates a lupus anticoagulant is reported. The presence of a circulating anticoagulant adds to the list of potential hemorrhagic diatheses in myelofibrosis and also demonstrates myelofibrosis to involve a system that may be separate from the myeloid elements of the bone marrow.
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keywords = bone
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2/129. Circulating levels of beta-chemokines in systemic lupus erythematosus.

    OBJECTIVE: Recent evidence suggests the role of beta-chemokines and their receptors in human immunodeficiency virus infection. We examined the serum levels of beta-chemokines in patients with systemic lupus erythematosus (SLE). methods: The serum levels of beta-chemokines, macrophage inflammatory protein-1alpha (MIP-1alpha), MIP-1beta, RANTES, and monocyte chemoattractant protein-1 (MCP-1) in patients with SLE were determined by ELISA. RESULTS: There were significant differences between the patients with SLE and healthy controls in the serum concentrations of RANTES (p < 0.001) and MCP-1 (p < 0.01), but not MIP-1alpha (p = 0.07) and MIP-1beta (p = 0.68). A decrease of RANTES and an increase of MCP-1 was observed with the progression of disease activity in the patients with SLE. CONCLUSION: Changes in the serum levels of RANTES and MCP-1 may indicate an interaction between SLE disease activity and the production of beta-chemokines.
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ranking = 125.93152250142
keywords = macrophage
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3/129. Study of systemic lupus erythematosus in temporal bones.

    Despite some reports of sensorineural hearing loss with systemic lupus erythematosus (SLE), its pathologic correlate has remained unidentified due to the scarcity of human temporal bone studies. We here present findings in 14 temporal bones from 7 patients with SLE, examined histologically and immunohistochemically for pathologic conditions in the cochlea that might relate to their otologic histories. Blue-staining concretions were seen in the stria vascularis of 6 ears. Most of the cases showed a loss of spiral ganglion cells, with various degrees of hair cell loss and atrophy of the stria vascularis. One ear demonstrated formation of fibrous tissue and bone throughout the cochlea, with complete loss of the membranous labyrinth. Cochlear hydrops was found in only 1 ear. These findings in temporal bones from patients with SLE are discussed in relation to autoimmune disease of the inner ear.
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ranking = 8
keywords = bone
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4/129. Significance of serum c-reactive protein elevation in patients with systemic lupus erythematosus.

    serum c-reactive protein (CRP) concentration was determined by a radial immunodiffusion method in serum samples collected over a mean period of 19 months from a group of 17 patients with systemic lupus erythematosus (SLE). Thirty-two episodes of CRP elevation were detected. Twenty of these were associated with active SLE, and 9 with proven or possible infection or bone fracture. In 3 cases no explanation for CRP elevation was readily apparent. There were 8 instances of onset or exacerbation of lupus activity without accompanying CRP elevation. These data indicate that CRP elevation in the course of SLE is frequently associated with activation of lupus, and that detection of such elevation does not differentiate between lupus activity and infection.
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5/129. Treatment of severe neutropenia due to Felty's syndrome or systemic lupus erythematosus with granulocyte colony-stimulating factor.

    OBJECTIVES: To examine the efficacy and safety of recombinant human granulocyte colony-stimulating factor (rhG-CSF) and recombinant human granulocyte-macrophage colony-stimulating factor (rhGM-CSF) for the treatment of severe neutropenia due to Felty's syndrome (FS) or systemic lupus erythematosus (SLE). methods: Eight patients with absolute neutrophil counts (ANC) below 1,000/microL attributable to FS (n = 4) or SLE (n = 4) were treated with rhG-CSF. The hematologic and clinical response as well as side effects were recorded. In addition, reports on the use of rhG-CSF/rhGM-CSF in FS and SLE retrieved from the English language literature were analyzed. RESULTS: RhG-CSF effectively corrected neutropenia due to FS and SLE in seven of the current eight patients. In 54 of 55 FS and SLE patients retrieved from the literature, G-CSF or GM-CSF, respectively, proved to be effective at elevating the neutrophil count, which was often associated with improvement of infectious complications. The neutrophil count often declined again when growth factor treatment was stopped but generally stabilized at a level that exceeded the pretreatment count. Side effects included rare cases of thrombocytopenia, arthralgias, and development of cutaneous leukocytoclastic vasculitis. Side effects were dose dependent and resolved when treatment was discontinued. One of our own patients and 17 previously reported patients continued to benefit from long-term administration of rhG-CSF over periods of more than 40 months. CONCLUSIONS: RhG-CSF is an effective and generally well-tolerated treatment for neutropenia due to FS or SLE. Exacerbation of the underlying rheumatic condition due to G-CSF appears to be rare if G-CSF is administered at the lowest dose effective at elevating the ANC above 1,000/microL.
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ranking = 125.93152250142
keywords = macrophage
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6/129. Multiple fish vertebra deformity in child with systemic lupus erythematosus: a case report.

    We report an 11-year-old female patient with multiple fish vertebra deformity, which occurred in the course of treatment with corticosteroids for systemic lupus erythematosus (SLE). She was treated for SLE with predonisolone (30 mg per day) from April 2, 1996, and presented at our outpatient clinic for an osteoporosis check-up on April 27. She was 132 cm tall with-1.7 standard deviation of the average height, and X-ray examination revealed no evidence of osteoporosis in the spine. Bone mineral density (BMD) was 74.7% of the average BMD. Subsequently, she grew to 136 cm in September. However she began to have low back pain (LBP) from November, and received alfacalcidol. LBP deteriorated after pulse therapy with methylpredonisolone. In June 1997, X-ray examination revealed multiple fish vertebra deformity with 58.3% of the average BMD. Moreover her height had decreased to 131cm. She underwent combination therapy with elcatonin and alfacalcidol. In September 1999, she had no LBP nor progression of fish vertebra deformity. However she had no growth in height. Corticoseroids and SLE have multiple effects on bone metabolism, making the treatment of porosis complicated and difficult.
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keywords = bone
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7/129. An unusual case of systemic lupus erythematosus, lupus nephritis, and transient monoclonal gammopathy.

    A 23-year-old female patient suffering from active systemic lupus erythematosus (SLE) was treated with azathioprine (2 mg/kg per day) and prednisone. lupus nephritis class III with increasing proteinuria developed 28 months after disease onset. Treatment was switched to monthly pulse cyclophosphamide administered intravenously for 6 months (total dose 6.3 g), followed by oral azathioprine and low-dose prednisone to maintain partial remission. Eight months later, the patient developed an acute exacerbation of SLE with fever, proteinuria of 9.1 g/day, pancytopenia, and cerebral involvement with cephalgias and a grand mal seizure. She responded well to high-dose corticosteroids (500 mg prednisolone pulses over 3 days, i.v.) and was azathioprine switched from to methotrexate (12.5-15 mg per week). Under this treatment, lupus activity gradually decreased and the patient felt well again. Five years after the initial diagnosis of SLE, a rapidly increasing immunoglobulin g-kappa type (IgG-kappa) monoclonal gammopathy developed, reaching a maximal serum paraprotein concentration of 73.5 g/l. Bone marrow biopsy revealed 15% of moderately abnormal, highly differentiated plasma cells arranged in small clusters and expressing IgG-kappa. No bony lesions were detectable on skeletal radiographs. Pulses of dexamethasone (40 mg) were administered and led to a transient decrease of paraproteinemia to a minimum of 31.9 g/l, followed by an increase to 62 g/l. At that point, high-dose chemotherapy supported by autologous stem cell transplantation was considered. Due to an intermittent pneumococcal septicemia, methotrexate was discontinued and dexamethasone was replaced by 5-10 mg cloprednol. At this point, totally unexpectedly, the paraprotein decreased spontaneously without any further cytostatic treatment and was no longer detectable 1 year later. Concomitantly, plasma cell counts in bone marrow biopsies fell to below 5%. As SLE remained inactive, the patient became pregnant and gave birth to a healthy child. During late pregnancy, SLE activity flared up with rising proteinuria and blood pressure. Therefore, after delivery, cyclophosphamide (100 mg/day, orally) was readministered for 4 months, resulting in an improvement of kidney function with stable proteinuria of 1-2 g/l to date. paraproteins are no longer detectable. In conclusion, this case report documents the rare event of transient paraproteinemia in a patient with SLE. A self-limiting regulatory defect in the control of a terminally differentiated B-cell clone may be the origin of this phenomenon.
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keywords = bone
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8/129. Severe hypercalcemia and systemic lupus erythematosus.

    A rare case of severe hypercalcemia strongly associated with Systemic Lupus Erythematosus (SLE) is reported. On admission, a young woman showed severe hypercalcemia and photosensitivity. Criteria for diagnosis of SLE were not sufficient. All causes, common and uncommon, of hypercalcemia were excluded. Radiographs of the skeleton were normal. One year later diagnosis of SLE was evident. In addition, diffuse and severe osteopenia and chest deformities had occurred. The treatment of SLE normalized persistently calcemia. Mild elevation of calcium levels occurred during flares of SLE. It has been hypothesized that hypercalcemia in patients with SLE could be caused by the presence of stimulatory anti-PTH receptor antibodies. This case report suggests that in patients with severe hypercalcemia associated with SLE early diagnosis and treatment of SLE may prevent bone loss. In these patients the prevention of severe bone damage is very important. Indeed, severe osteopenia may favour skeletal deformities and fractures; in addition it may represent a serious obstacle in using adequate doses of glucocorticoids for treatment of SLE.
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ranking = 2
keywords = bone
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9/129. Bilateral femoral osteomyelitis with knee arthritis due to salmonella enteritidis in a patient with systemic lupus erythematosus.

    A bilateral knee septic arthritis due to salmonella enteritidis developed in a female patient affected by long-standing systemic lupus erythematosus (SLE) with cardiac and renal involvement treated with immunosuppressants and corticosteroids. Because avascular necrosis and multiple osteomyelitic areas were detected at the same time in both right and left femoral condyles, an early localisation of Salmonella into the bone was assumed. Involvement of the joints was regarded as consequence of local dissemination of infection. Ampicilline (0.2 g/kg body weight daily for 2 months) plus ciprofloxacin (1.5 g daily for 12 months) and withdrawal of immunosuppressants appeared to be effective in preventing complications of infection.
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ranking = 1
keywords = bone
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10/129. Myelofibrosis in systemic lupus erythematosus.

    In this study we present a case of coexisting systemic lupus erythematosus (SLE) and myelofibrosis. literature review supports the fact that the two diseases rarely occur together in the same patient. The young female patient studied was admitted with pancytopenia and a clinical picture which met the criteria of SLE. Histological examination of the bone marrow biopsy revealed severe myelofibrosis with hypocellularity of the myeloid cell lines. Treatment with immunosuppressive and colony stimulating factor led to slow but complete regeneration of the bone marrow and subsequently to an improved haematological status, and the patient was spared bone marrow transplantation.
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