Cases reported "Osteomyelitis"

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1/18. 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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2/18. Kawasaki syndrome-like illness associated with infection caused by enterotoxin B-secreting staphylococcus aureus.

    Two children had symptoms and clinical signs that were characteristic of the diagnostic criteria for Kawasaki syndrome, temporally associated with staphylococcus aureus bacteremia. One child initially had focal osteomyelitis that was evident clinically and radiographically, and radiographic evidence of multifocal osteomyelitis was noted at follow-up. The blood-borne S. aureus isolates from these two patients secreted staphylococcal enterotoxin B and were negative for toxic shock syndrome toxin. Staphylococcal and streptococcal superantigens may play a role in the pathogenesis of some cases of Kawasaki syndrome or Kawasaki syndrome-like illness.
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3/18. Successful unrelated bone marrow transplantation for a patient with chronic granulomatous disease and associated resistant pneumonitis and aspergillus osteomyelitis.

    We describe the successful treatment of a 20-year-old patient with chronic granulomatous disease (CGD), by unrelated bone marrow transplantation (UBMT). The patient is relatively old compared to other CGD patients treated with BMT. He had had repeated serious infections from early childhood and was diagnosed as CGD, gp91-phox deficiency. Prolonged antibiotic-resistant pneumonitis worsened when the patient was 18 years old. In addition, he suffered aspergillus osteomyelitis and acute renal failure due to amphotericin b. He received 94 granulocyte transfusions from 94 adult donors and the infections gradually improved. In September 1998, at 20 years of age, he underwent UBMT from an HLA 6 antigen-matched male donor, with CY and TBI conditioning. He received MTX and CsA as prophylaxis against GVHD. No serious complications occurred and rapid engraftment was achieved. Acute GVHD (grade 2, at day 19) and chronic GVHD (limited, at day 192) occurred. However, both were easily controlled. The patient is alive and well with no late rejection 26 months after UBMT.
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4/18. arizona hinshawii osteomyelitis with antecedent enteric fever and sepsis. A case report with a review of the literature.

    A case of arizona osteomyelitis of the spine which occurred 11 months after an episode of gastroenteritis and enteric fever is presented. As close biochemical and antigenic relative of salmonella, arizona infection produces a similar clinical course with gastrointestinal manifestations frequently preceding localized infections by several months. The boney lesion in the present case and in three of the four other cases of arizona osteomyelitis described in the literature was a chronic inflammation which may have a xanthomatous component. The bone destruction caused by arizona infection is less severe than that of tuberculosis or pyogenic osteomyelitis. Proposed treatment of arizona osteomyelitis consists of debridement of the localized infection and long term antimicrobial therapy.
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5/18. Cryptococcal osteomyelitis and cellular immunodeficiency associated with interleukin-2 deficiency.

    We describe an unusual example of cellular immunodeficiency associated with interleukin-2 deficiency in an otherwise healthy 15-year-old boy who had isolated cryptococcal osteomyelitis of the scapula at 10 years of age. His previous medical history was remarkable only for prolonged, severe varicella infection at 6 years of age. He had persistent moderate lymphopenia, anergy, and absent lymphocyte blastogenic responses to mitogens, antigens, or monoclonal T cell antibodies. Subnormal blastogenic responses were seen after exposure to high concentrations of phorbol esters. Immunoglobulin levels and specific antibodies were normal. The patient has been in good health since treatment of his osteomyelitis. However, his lymphocyte blastogenic responses to mitogens have remained absent during 4 years of observation; investigation of the cause revealed a specific interleukin-2 deficiency resulting from defective generation of interleukin-2 messenger ribonucleic acid. Secretion of interleukin-1 by monocytes was normal, suggesting that the abnormal blastogenic response and interleukin-2 production were due to a problem intrinsic to T lymphocytes. The generation of messenger ribonucleic acid for interleukin-4 was not affected. interferon-gamma was produced at subnormal levels. The addition of recombinant interleukin-2 restored lymphocyte blastogenic responses and increased the expression of interleukin-2 receptors. The clinical findings and immunologic abnormalities present in this patient differ from other primary and secondary immunodeficiencies associated with interleukin-2 deficiency. Thus our observations in this patient extend the spectrum of immunodeficiencies associated with abnormalities in the production of this important cytokine.
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6/18. brucellosis: an unusual diagnosis for a seronegative patient with abscesses, osteomyelitis, and ulcerative colitis.

    A 16-year-old girl developed multiple subcutaneous abscesses, osteomyelitis, and severe colitis. On the patient's second admission, a single blood culture--and, subsequently, a specimen of pus--yielded brucella melitensis biovar 1. A second set of serologic tests, including the rose bengal test, the standard tube agglutination test, the CF test, and Coombs' test, were all negative for Brucella on the patient's second admission and 1 month later. However, a lymphocyte proliferation assay with extracted antigen of Brucella was markedly positive. Thus, this case illustrates that patients with B. melitensis infection may have a unique clinical presentation and that the lymphocyte proliferation assay is an important diagnostic tool for patients whose serologic test results are negative but for whom brucellosis is suspected.
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7/18. Candidal infection of bone. Assessment of serologic tests in diagnosis and management.

    In this case report, 30 sera from a 25-year-old heroin abuser with intervertebral candidosis were treated for the presence of anti-candida albicans antibodies by agglutination, counterimmunoelectrophoresis, and indirect immunofluorescent assay. Sera were also adsorbed with heat-killed blastospores to remove antibodies against yeast-phase cells and tested by indirect immunofluorescent assay for anti-C. albicans germ tube antibodies (CAGTAs). Humoral responses to candidal 47-kD antigen were studied by immunoblotting in 23 unadsorbed sera. Anti-C. albicans antibodies were found in high titers by the three procedures but correlated poorly with the clinical evolution of the disease. CAGTAs were present from the beginning of the infection: Titers decreased in association with antifungal treatment and the patient's improvement, eventually becoming negative. Only class IgG antibodies to the 47-kD antigen were detected. These were present during the full course of the infection, failing to disappear at the end of the study. In this case, detection of CAGTAs appeared to be an important aid to diagnosis of the bony candidal infection, as they are detected early during the illness and seemed to have a prognostic significance.
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8/18. Osteomyelosclerosis with granulocytic sarcoma of chest wall. Morphological, ultrastructural, immunologic, and cytogenetic study.

    A case of granulocytic sarcoma presenting as a soft-tissue tumor in the chest wall in a patient with osteomyelosclerosis is reported. The tumor mass was detected by a computed tomographic scan during an investigation of the cause of chest pain in a 58-year-old man. biopsy of the mass showed findings compatible with either a large-cell lymphoma or a granulocytic sarcoma. The latter was confirmed by naphthol-ASD-chloracetate esterase stain and electron microscopic examination. Immunologic study of the tumor mass showed expressions of membrane/cytoplasmic CD 13 and CD 15 antigens. In addition, the tumor cells coexpressed CD 19, although all other T- and B-cell-associated antigens were absent. Cytogenetic study showed translocation t(1;7)(q11;q11) with a net deletion of the entire long arm of chromosome 7 and duplication of the long arm of chromosome 1. Peripheral blood examination showed typical leukoerythroblastosis with teardrop poikilocytosis, large hypogranular platelets, and 0.11 myeloblasts. A bilateral iliac bone marrow biopsy at this time showed osteomyelosclerosis. The patient was treated with hydroxyurea followed by local irradiation, resulting in marked reduction in the size of the tumor and in the pain. He was asymptomatic without any progression in hematologic parameters 10 months after the initial diagnosis.
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9/18. The immunochemical characterisation of circulating immune complex constituents in candida albicans osteomyelitis by isoelectric focusing, immunoblot, and immunoprint.

    Circulating immune complexes present in the serum of a patient with systemic lupus erythematosus, end-stage renal failure, and thoracic vertebral candida albicans osteomyelitis were sequentially analysed by isoelectric focusing, immunoblotting, and immunoprinting. Candida antigens (including mannoproteins), Clq and C3 complement components, and specific anti-Candida antibody were detected within polyethylene glycol precipitated complexes. An antigen of 47K molecular weight was amongst those demonstrated by polyacrylamide gel electrophoresis to be present within the complexes. It has been proposed elsewhere that a serologic response to a 47K protein is predictive of recovery from candida albicans infection. Free antibody had specificity for Candida antigens ranging in molecular weight from 18K to more than 100K including a 47K component. The analytical techniques employed allowed rapid and precise identification of the components of one particular immune complex system and will be widely applicable to the dissection of other diverse systems.
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10/18. candida albicans vertebral osteomyelitis in chronic renal failure.

    Invasive candidal infections are encountered with increasing frequency in compromised hosts but bone infection is uncommon. A woman with systemic lupus erythematosus and end-stage renal failure managed by continuous ambulatory peritoneal dialysis developed a painful thoracic kyphosis and a lytic lesion in the vertebral bodies of T10 and T11. blood cultures were sterile but bone biopsy material contained candida albicans which also grew on culture. Circulating immune complexes were measured in high levels and contained candida antigens and specific anti-candida antibody as determined by isoelectric focusing, immunoblotting and immunoprinting techniques. Pain persisted after anti-fungal therapy had sterilized the lesion necessitating surgical excision of affected vertebrae, kyphosis correction and iliac crest bone grafting. The titres of circulating immune complexes and anti-candidal precipitins closely paralleled the clinical course.
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