A case where a breast tumor was the initial manifestation of Wegener's granulomatosis is presented. Breast biopsy revealed granulomatous mastitis, and treatment with tuberculostatic drugs was started. Following an initial improvement the patient finally died. Postmortem, the diagnosis of generalised Wegener's granulomatosis was made. Although Wegener's granulomatosis is a very rare cause of granulomatous mastitis, it should be considered in the differential diagnosis. A brief outline of the literature is given.
The optimal treatment for severe subglottic stenosis secondary to Wegener's granulomatosis remains controversial. We report the case of a symptomatic middle-aged woman who was successfully treated with intratracheal dilation and intralesional injection of corticosteroids. The literature related to this issue is being reviewed.
We present the case of a 69 yr old, white male, suffering from diffuse interstitial lung disease, finally diagnosed as paracoccidioidomycosis or South American blastomycosis. During the course of his disease, antineutrophil cytoplasmic antibodies (c-ANCAs) became positive, suggesting the possibility of a Wegener's granulomatosis.Transbronchial biopsies and a video-assisted thoracoscopic lung biopsy revealed only the pulmonary yeast infection, without other co-existing pathology. During treatment with itraconazole, the patient improved clinically and functionally, and c-ANCAs became negative. Serological monitoring confirmed the diagnosis.To our knowledge, this is the first report describing positive c-ANCAs in a patient with paracoccidioidomycosis. It re-emphasizes the fact that cautious interpretation of c-ANCAs in patients without convincing clinical signs or pathological evidence of a granulomatous vasculitis is absolutely necessary. In this era of increased mobility, a thorough medical history, including documentation of travel, remains an inexpensive tool in making a diagnosis and is still the cornerstone of good medical practice. Eur Respir J 1997; 10: 2419-2422 Case reportA 69 yr old, white male, who underwent coronary artery bypass surgery in 1983, presented with a history of progressive dyspnoea on exertion, chest tightness and a nonproductive cough of 4 month duration. Previous treatment with erythromycin had not improved symptoms. He had a smoking history of 45 pack-years.From 1948 to 1963, the patient worked on a plantation in Paraguay and travelled in Brasil and Argentina. Afterwards, he worked as a representative for laboratory equipment in Belgium. He denied asbestos exposure. His medication consisted of dipyridamole and acetylsalicylic acid.Physical examination was completely normal. Chest radiography and computed tomography (CT) of the thorax revealed diffuse alveolar filling and a reticulonodular interstitial pattern ( fig. 1). Laboratory examination only demonstrated a polyclonal hypergammaglobulinaemia. Pulmonary function tests showed a moderately restrictive ventilatory pattern, with a vital capacity (VC) of 2.2 L (61% of predicted value), a forced expiratory volume in one second (FEV1) of 1.7 L (61% pred), a total lung capacity (TLC) of 4.8 L (74% pred), and an abnormal carbon monoxide transfer factor (TL,CO) of 40% pred. There was a mild hypoxaemia, with an arterial oxygen tension (Pa,O 2 ) of 9.6 kPa. Bronchoscopy showed no endobronchial abnormalities. Cultures of bronchoalveolar lavage fluid remained sterile and a cell count revealed elevated numbers of lymphocytes (21%), eosinophils (4.5%) and neutrophils (6.5%). The CD4/CD8 ratio was normal. Transbronchial biopsies in the left lower lobe showed normal alveolar tissue.In the absence of a clear diagnosis, empirical therapy with methylprednisolone at a dose of 1 mg·kg -1 was initiated. After initial clinical, radiological and lung functional improvement, the patient was hospitalized because of severe dyspnoea and temperature up...
Twelve patients suffering from partially reversible chronic obstructive pulmonary disease (COPD) took past in a single blind, randomised, 4-way cross-over trial to determine the optimal dose and duration of action of the anticholinergic agent oxitropium bromide (OTB) inhaled as a nebulised solution. Single doses of 500, 1000, 1500 and 2000 micrograms nebulised OTB were compared during a 6 hour-observation period. Lung function test results indicated that 500 and 1000 micrograms OTB only induced slight bronchodilatation, whereas 1500 and 2000 micrograms OTB produced a significantly greater increase in mean FEV1 compared to 500 micrograms. There was a trend for 2000 micrograms to be superior to 1000 micrograms, but 2000 micrograms and 1500 micrograms were not significantly different. Significant bronchodilatation (> 15% rise in FEV1 from baseline) persisted for 6 h after 1500 micrograms. A significant decrease in airway resistance (Raw) was observed following inhalation of 2000 micrograms. The mean decrease in Raw was 33% after 30 min, 20% after 4 h and 12% after 6 h. In this trial, 2000 micrograms OTB administered by an ultrasonic nebuliser was the optimal dose, but a satisfactory result was also obtained with 1500 micrograms.
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