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1/5. ecthyma gangrenosum and septic shock syndrome secondary to chromobacterium violaceum.

    chromobacterium violaceum is a gram-negative bacterium of soil and water in tropical and subtropical environments. Typically, it is considered a bacterium of low virulence although, uncommonly, it causes human infection, particularly in persons with defects in host defenses. Infection generally follows exposure of broken skin to contaminated water and soil, and is often characterized by pustules, lymphadenitis, fever, and vomiting, as well as rapid dissemination and a high mortality rate. Unfortunately, because C violaceum is ubiquitous, it is often dismissed as a contaminant when cultured. Because rapid diagnosis (by taking appropriate specimens) and treatment are vital to a good prognosis, it is imperative that physicians be aware of this organism. In addition, patients with chromobacterial infections should have an immunologic workup because infections in immunocompetent individuals are rare. Here we report an aggressive yet nonfatal case of C violaceum septicemia in an adolescent male, diagnosed through a punch biopsy of a skin lesion, and resulting in a new diagnosis of chronic granulomatous disease.
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2/5. natural history of chronic granulomatous disease.

    An analysis of the patient characteristics, clinical course, and therapy of 52 patients with chronic granulomatous disease (CGD) was carried out through a multinstitutional questionnaire survey in order to further define the natural history of this disorder of phagocyte microbicidal function. The results suggest that CGD is characterized by significant heterogeneity in clinical course and prognosis. The age at diagnosis, severity of disease, or early death in an affected sibling does not seem to be of value in predicting the prognosis for individual patients with CGD. A more detailed, large-scale multicenter study and the establishment of a patient registry is recommended.
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3/5. Necrotizing sarcoid granulomatosis with suppurative features.

    Necrotizing sarcoid granulomatosis has become a well-defined entity within the spectrum of disorders classified as pulmonary granulomatosis with angiitis. It is characterized clinically by disease generally restricted to the chest, steroid sensitivity, and a good prognosis. Pathologically, confluent granulomas, vasculitis, and bland necrosis are seen. The authors report a patient initially presenting with the typical clinical and pathologic features of necrotizing sarcoid granulomatosis who, on subsequent recurrence, demonstrated a suppurative character to the necrosis. This feature has not been reported previously, and its recognition will allow more cases to be diagnosed correctly. serum angiotensin converting enzyme (ACE) was not elevated, and ACE could not be demonstrated in tissue from the lung biopsy obtained during recurrence of disease. This further suggests significant differences between this entity and sarcoid.
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4/5. Chronic granulomatous disease of childhood: a changing pattern?

    We have followed nine male patients with Chronic Granulomatous disease at The Hospital for Sick Children, Toronto, since 1972. The diagnosis was established in each case by the failure of neutrophils to reduce nitroblue tetrazolium dye and to kill staphylococcus aureus normally in vitro. bacterial infections began between 6 months and 14 years of age. In five of the nine patients, infections began after 4 years of age. The first significant infection in five patients was a liver abscess(es), and one patient each had lymphadenitis, pulmonary aspergillosis, a parapharyngeal abscess, and a draining inguinal incision following surgery. Following diagnosis, all patients were started on trimethoprim-sulfamethoxazole at a dose of 2 mg/kg/day of trimethoprim. The patients have been followed for 50 patient-years. Five of nine patients have been free of infection during 16 years of observation. For the remaining four patients, there have been six infections during 34 years of observation. A possible infection-related death occurred in one patient. The patients reported here appear to differ from those in previous reports. They present later in life, often with a liver abscess. They have a low incidence of subsequent bacterial infections which may, in part, be due to trimethoprim-sulfamethoxazole prophylaxis. The patients with chronic granulomatous disease reported here appear to have a better prognosis than previously thought.
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5/5. invasive pulmonary aspergillosis in chronic granulomatous disease: response to systemic prednisolone treatment and locally applied amphotericin b.

    A five-year-old boy with deforming and destructive invasion of pulmonary aspergillosis to the thoracic cage was diagnosed as having chronic granulomatous disease. Conventional antifungal therapy failed in this patient. prednisolone therapy was added and rapidly improved the general condition of patient but deterioration had already been very rapid. The failure of systemic therapy prompted us to give amphotericin b locally to the granulomatous lesions through the bronchocutaneous fistula. This application yielded a good clinical response with closure of fistula. Despite this improvement the patient died from septicemia. We believe that systemic prednisolone treatment is useless in such cases, but local application of amphotericin b into the granulomatous lesions together with systemic therapy during the earlier stages of infection can contribute to a change in prognosis.
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