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1/296. Pseudogaucher cells in cutaneous Mycobacterium avium intracellulare infection: report of a case.

    We report on a patient infected with human immunodeficiency virus, and with cutaneous Mycobacterium avium intracellulare, in whom many cells with abundant reticulated cytoplasm resembling the characteristic cells of Gauchers disease ("pseudogaucher cells") were noted within the dermal infiltrate on biopsy. Although pseudogaucher cells have been reported in association with M. avium intracellulare infection in extracutaneous sites, this is, to our knowledge, the first report of cutaneous pseudogaucher cells in the skin. ( info)

2/296. Management of opportunistic infections in acquired immunodeficiency syndrome. I. Treatment.

    A case report of a patient infected with human immunodeficiency virus (HIV) is described. The patient presents with a multitude of medical complaints that are of acute or subacute onset. The medical examination of these complaints is described and includes algorithms for the diagnosis and treatment of the most common HIV-related opportunistic infections, including pneumocystis carinii pneumonia, toxoplasmosis, mycobacterium avium complex, cytomegalovirus infection, and cryptococcal meningitis. ( info)

3/296. Immunopathology as a result of highly active antiretroviral therapy in hiv-1-infected patients.

    OBJECTIVE: Unusual clinical inflammatory syndromes associated with underlying previously unrecognized opportunistic infections are increasingly being noted shortly after starting highly active antiretroviral therapy (HAART). This study examined the possible relationship between such unexpected disease manifestations and in vitro parameters of microbial antigen-specific immune reactivity in patients infected with hiv-1 who had a Mycobacterium avium intracellulare or mycobacterium xenopi infection. DESIGN: in vitro T-cell proliferation experiments were performed after specific stimulation of a patient's peripheral blood mononuclear cells (PBMC) with M. avium and M. xenopi antigen and non-specific stimulation with phytohaemagglutinin (PHA). The results were compared with appropriate controls. patients: Five patients who presented with unusual clinical syndromes associated with M. avium or M. xenopi infection within weeks of experiencing large rises in CD4 cell counts following the initiation of antiretroviral therapy. RESULTS: In all patients except one, mycobacteria-specific lymphoproliferative responses rose significantly following HAART; this was temporally associated with elevations in CD4 cell counts and the occurrence of clinical disease. The patient with M. xenopi infection appeared to clear his infection subsequently without antimycobacterial therapy. In three of the four patients with M. avium infection, antimycobacterial treatment could be stopped without recurrence of infection. CONCLUSION: Our findings support the hypothesis that HAART may lead to clinically relevant inflammation as a result of restoration of specific immune reactivity against microbial pathogens that are subclinically present at the time treatment is initiated. Continuation of HAART may subsequently result in protective immunity and clearance of infection. ( info)

4/296. Disseminated mycobacterium avium complex infection presenting as osteomyelitis in a normal host.

    Disseminated mycobacterium avium complex (MAC) infection presenting as a painful lytic femur lesion with associated fever, night sweats and weight loss occurred in a 45-y-old woman with apparent normal immune function. Surgical drainage and 24 months of medical therapy resulted in a cure. ( info)

5/296. Polyarthralgia-arthritis syndrome induced by low doses of rifabutin.

    We describe 2 cases of polyarthralgia-arthritis syndrome induced by rifabutin, an effective treatment for infections of Mycobacterium avium intracellulare complex. This syndrome has been reported with doses higher than 1 g per day when rifabutin is given in monotherapy. But our cases were treated with low doses, 300-450 mg per day, in combination with clarithromycin. The plasma concentration of rifabutin has been shown to be increased by clarithromycin, suggesting that co-prescription of clarithromycin could lead to development of rifabutin induced polyarthralgia-arthritis syndrome. ( info)

6/296. Infection by rhodococcus equi in a patient with AIDS: histological appearance mimicking Whipple's disease and mycobacterium avium-intracellulare infection.

    rhodococcus equi pneumonia with systemic dissemination is being reported increasingly in immunocompromised patients. This is the first case report of disseminated R equi infection with biopsy documented involvement of the large intestine. The patient was a 46 year old male with AIDS who was diagnosed with cavitating pneumonia involving the left lower lobe. R equi was isolated in culture from the blood and lung biopsies. Subsequently, the patient developed anaemia, diarrhoea, and occult blood in the stool. colonoscopy revealed several colonic polyps. Histological examination of the colon biopsies showed extensive submucosal histiocytic infiltration with numerous Gram positive coccobacilli and PAS positive material in the histiocytes. Electron microscopy showed variably shaped intrahistiocytic organisms which were morphologically consistent with R equi in the specimen. Disseminated R equi infection may involve the lower gastrointestinal tract and produce inflammatory polyps with foamy macrophages which histologically resemble those seen in Whipple's disease and mycobacterium avium-intracellulare infection. ( info)

7/296. Successful treatment of chylous ascites secondary to mycobacterium avium complex in a patient with the acquired immune deficiency syndrome.

    chylous ascites is a rare form of ascites, the presence of which generally denotes a very poor long term prognosis. We report the case of a patient with acquired immune deficiency syndrome (AIDS) and massive chylous ascites secondary to mycobacterium avium complex (MAC) infection, identified in the ascitic fluid by a dna probe assay. With multidrug anti-MAC therapy the ascites resolved completely, and the patient has survived for >21 months. diagnosis and treatment of MAC-related chylous ascites are reviewed. ( info)

8/296. Potential association between calcified thoracic lymphadenopathy due to previous histoplasma capsulatum infection and pulmonary mycobacterium avium complex disease.

    BACKGROUND: Among patients with pulmonary disease due to mycobacterium avium complex (MAC) seen recently at our center, a substantial number have had extensive calcified mediastinal, hilar, and peribronchial lymphadenopathy, a finding historically inconsistent with pulmonary MAC disease. METHOD: We retrospectively studied the frequency of calcified lymphadenopathy in the chest and prevalence of known risk factors for MAC infection in 79 patients with pulmonary MAC disease who were referred to our hospital over a 1-year period. RESULTS: Calcified intrathoracic adenopathy was present in 25 of the 79 patients (32%). Residential histories revealed that 20 of the 25 patients (80%) with such calcified chest adenopathy reported living for substantial periods in the regions indigenous for histoplasma capsulatum. In contrast, the residences of patients without calcified chest adenopathy were more evenly distributed throughout the country. Nineteen of these 25 patients (76%) with calcified chest adenopathy had no known predisposing risk factor for the infection; in contrast, the proportion of patients with no calcified adenopathy who also had no identifiable classic risk factor tended to be lower (32/54, 59%). CONCLUSION: In this retrospective study, we observed that (1) a large number of patients with pulmonary MAC disease had no identifiable risk factor, (2) calcified chest adenopathy was present in one third of the patients, (3) the residential history of those with calcified adenopathy mirrored the endemic region of histoplasmosis, and, (4) conversely, those patients with pulmonary MAC who lived outside the histoplasmosis belt had no such adenopathy. Thus, we hypothesize that previous fungal infection may predispose the lungs of certain patients to subsequent invasion by MAC, presumably by airway distortion and/or parenchymal damage. ( info)

9/296. coinfection of visceral leishmaniasis and Mycobacterium in a patient with acquired immunodeficiency syndrome.

    We report a case of coinfection of visceral leishmaniasis and Mycobacterium avium-intracellulare in the same lesions in the small bowel and bone marrow of a 33-year-old man with acquired immunodeficiency syndrome who complained of abdominal pain and chronic diarrhea. The duodenal mucosa and bone marrow biopsy specimens showed numerous foamy macrophages packed with two forms of microorganisms that were identified histologically and ultrastructurally as Leishmania and Mycobacterium species. Visceral leishmaniasis is rarely suspected in patients residing in nonendemic countries including the united states. It should be included in the differential diagnosis for opportunistic infection in patients with acquired immunodeficiency syndrome. An appropriate travel history is important. To our knowledge, this is the first reported case showing coinfection of visceral leishmaniasis and Mycobacterium avium-intracelluulare in the same lesion in a patient with acquired immunodeficiency syndrome. ( info)

10/296. listeria monocytogenes and recurrent mycobacterial infections in a child with complete interferon-gamma-receptor (IFNgammaR1) deficiency: mutational analysis and evaluation of therapeutic options.

    We describe the history of a girl with interferon-gamma-receptor (IFNgammaR1) deficiency and studies performed to identify the molecular and clinical characteristics of this recently discovered disorder. This is the first report of a child from Northern europe with IFNgammaR1 deficiency. The patient, now 7 years old, first presented with swelling and reddening at the Bacille Calmette-Guerin (BCG) vaccination site, swelling of lymph nodes, hepatomegaly, and an unusually severe varicella rash at the age of 4 months. At that time, she was diagnosed with BCG histiocytosis without typical granuloma formation and was treated with antituberculous agents. During the clinical course of her illness, several different types of atypical mycobacteria and (for the first time in an IFNgammaR1-deficient patient) listeria monocytogenes were detected. Flow cytometric analysis showed that the patient's monocytes could not bind a monoclonal antibody specific for the IFNgamma-receptor. Our analysis of mRNA derived from the alpha-chain (IFNgammaR1) gene of this receptor revealed deletions of 173 bp and 4 bp in cDNA sequences originating from individual alleles. The 173 bp deletion was located between nucleotide positions 200 and 372, exactly matching those of exon 3, and the 4 bp deletion was located between nucleotide positions 561 and 564 of the coding region of the cDNA. Analysis of genomic dna revealed the presence of a G to T transition at the 5'end of the splice consensus sequence of intron 3, which explains the absence of exon 3. The other allele carried the 4-base-pair deletion (ACTC) at nucleotide positions 15-18 of exon 5. Twelve months after an allogeneic bone marrow transplantation, the patient had clinically improved. ( info)
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