A 68-year-old woman with a 2-month history of nausea and occasional vomiting was referred to the gastroenterologist. The patient complained of vague upper abdominal discomfort that was not influenced by meals. Laboratory findings were normal, and, specifically, no anemia was present (hemoglobin level, 8.4 mmol/L [normal, 7.1-9.9 mmol/L]; hematocrit level, 0.41 [normal, 0.34-0.48]). The patient reported normal bowel movements but mentioned a recent short period of diarrhea. Neither rectal bleeding nor melena was present, and fecal occult blood testing was not performed.A gastroscopy was performed by the gastroenterologist, who noted an extrinsic impression at the greater curvature of the stomach. The pyloric orifice was poorly recognized, and there was suspicion for an intussusception of part of the antrum into the duodenum. Rotation of the proximal part of the duodenum and reduction of the diameter of the duodenal lumen by more than 50% were present as the result of an external mass (Fig 1). The surface of the mass was mostly covered with normal-looking mucosa but became firm and ulcerative more distally. Because of the limited view at gastroscopy, it was difficult to determine whether the duodenal lumen reduction was the result of a submucosal mass or an external invading mass. Several biopsy specimens were obtained from both the normal-appearing mucosa proximal to the narrowed part of the duodenum and the more distal ulcerative surface.
Bronchocentric granulomatosis was first described by Liebow' and is a form of pulmonary granulomatosis and angiitis. The disease is characterised by its clinical and morphological features but the clinical2 and radiographic, manifestations are rather non-specific. Lung biopsy is necessary to establish the diagnosis and granulomatous inflammation, centred predominantly on the bronchi and bronchioles, is pathognomonic. According to the original description of Liebow,' one of the characteristics of bronchocentric grantilomatosis is the absence of extrapulmonary lesions. We here report a case of bronchocentric granulomatosis associated with seropositive polyarthritis. Case reportIn August 1981 a 58 year old man was admitted to another hospital with fever and an abnormal chest radiograph. He was treated initially with penicillin for suspected bacterial pneumonia; later doxycycline and erythromycin were given without definite improvement. When tomography subsequently showed enlarged hilar glands with persistent pulmonary shadowing, sarcoidosis was suspected. No biopsy was performed and prednisolone (20 mg daily) was started. After initial improvement, the clinical and radiographic manifestations worsened. In November 1981 he was referred to our hospital for further investigation. He was reported to have had mild asthmatic symptoms 20 years earlier, a myocardial infarction in 1975, and surgery for diverticulitis of the colon in early 1981.Shortly before transfer the patient developed polyarthritis. On admission he appeared ill, with a temperature of 390C, a persistent cough, haemoptysis, dyspnoea at rest, and chest pain. He had no history of Raynaud's phenomenon. On physical examination breath sounds were diminished over the lower part of the right lung and no wheezes were heard. The metacarpophalangeal and proximal interphalangeal joints were swollen and painful but there were no subcutaneous nodules. The chest radiograph (fig 1) showed bilateral macronodular lesions; the right lung, which showed cavitation, was more affected than the left. (fig 2) showed necrotising granulomatous inflammation centred predominantly on and destroying bronchioles. The adjacent pulmonary arteries showed vasculitis without evidence of bacteria or fungi. These features were consistent with bronchocentric granulomatosis. The dose of prednisolone was increased to 100 mg daily and the patient responded well. Aft r the dose had been tapered to 20 mg daily over an eight week period, the polyarthritis again worsened and the dose was increased temporarily to 30 mg.
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