A case of bronchocentric granulomatosis associated with uveitis]

A 32-year-old female was admitted to our hospital because of abnormal pulmonary shadows and a decrease in visual acuity. Analysis of peripheral blood revealed eosinophilia, and chest roentgenogram demonstrated multiple infiltrates in the right upper lung field. Pathological examination of transbronchial lung biopsy specimens revealed necrotizing granulomatous lesions in the walls of bronchioles, and a definitive diagnosis of bronchocentric granulomatosis was made. The cause of bronchocentric granulomatosis in this patient was suggested to be an allergic reaction to Aspergillus because of positive response to skin test for Aspergillus. Although it is reported that extrapulmonary involvement is rare in bronchocentric granulomatosis, the present case was associated with uveitis, and to our knowledge is the first reported case.     

Asbestosis,endobronchialAspergillus infection,and bronchocentric granulomatosis presenting with hemoptysis

A patient with pulmonary asbestosis developed hemoptysis and a lesion on chest radiograph secondary to bronchocentric granulomatosis (BG) associated withAspergillus fumigatus. Although pulmonaryAspergillus infection is a known complication of asbestos exposure, BG has not been previously reported in this disorder. This patient’s case further documents thatAspergillus hyphae may be present in BG unassociated with allergic bronchopulmonary aspergillosis. A wide range of processes affecting airways, including asbestos exposure, may be associated with bronchocentric granulomatosis.     

Corticosteroid monotherapy in a case of bronchocentric granulomatosis with a two-year follow-up

Bronchocentric granulomatosis (BCG) is a rare disease. Because of the possibility of fungal infection, BCG has usually been treated with corticosteroids and antifungal agent. However, fungi are not detected in all BCG tissues. We report a case of proven BCG by open lung biopsy without fungi in a woman with corticosteroids monotherapy and two-year follow-up.KEYWORDS : Bronchocentric granulomatosis (BCG), fungal infection, open lung biopsy, corticosteroids, allergic diseaseBronchocentric granulomatosis (BCG), which was first described by Liebow (1) in 1973, is a rare disease characterized by inflammation that begins within and is centered around the walls of bronchioles and manifests as fever, cough, wheezing, dyspnea, and blood eosinophilia. It is considered to occur primarily in asthmatic patients and to be closely related to fumigatus infection (2-4).Because of the possibility of fungal infection, BCG is usually treated with corticosteroids and an antifungal agent (5). However, fungi are not detected in all BCG tissues. The question thus arises of whether antifungal agents should be used in addition to corticosteroids in these patients. We here report a case of biopsy-proven BCG without accompanying fungal infection in a woman who was treated with corticosteroids alone and followed-up for two years.     

Pulmonary angitis and granulomatosis]

16 patients of Wegener's granulomatosis, 4 of allergic angiitis and granulomatosis, 3 of lymphomatoid granulomatosis and 1 of necrotizing sarcoid granulomatosis were reported. In this group of different diseases, characteristic pathological manifestations are inflammatory cellular infiltration of vessel wall combined with destruction and necrosis of pulmonary parenchyma. There was little difference in their clinical features. In most of the cases, fever and systemic symptoms related to lung and extrapulmonary organs were present. Correct diagnosis of these diseases is very important, because both the prognosis and therapy are different. As for the prognosis, it ranges from benign (necrotizing sarcoidal granulomatosis) to very malignant (lymphomatoid granulomatosis). The clinical features of each illness were reviewed with emphasis on their histopathologic findings. The therapeutic effect and final outcome were followed.     

A case of bronchocentric granulomatosis presenting as a tumorous shadow on chest X-ray film]

A 57-year-old woman was admitted to our hospital with cough, sputum and abnormal chest X-ray. In summer, 1989, she developed cough which gradually worsened in autumn. In November, the chest X-ray revealed a tumorous shadow in the left suprahilar region. On admission, there were no symptoms of bronchial asthma. Chest X-ray revealed a subpleural tumorous shadow in the left upper lung field. X-ray findings suggested that the tumorous shadow in the suprahilar region moved to the left peripheral upper lung field. Left B1+2 orifice obstruction with necrotic tissue was seen on fiberoptic bronchoscopy. Transbronchial biopsies failed to yield specific diagnostic findings, except for bronchitis with exudate containing eosinophils. In February, 1990, she developed hemosputum and left chest pain. Chest X-ray showed consolidation in the left apical lung field. Left upper lobectomy was performed. Histological examination disclosed many granulomas with central necrosis around the bronchi, and aspergillus hyphae were seen. These findings are compatible with bronchocentric granulomatosis without asthma.     

A case of Carrington's chronic eosinophilic pneumonia associated with bronchocentric granulomatosis in an asthmatic patient

A case of Carrington's chronic eosinophilic pneumonia associated with bronchocentric granulomatosis in a young asthmatic woman is reported. Among the numerous forms of "eosinophilic lung", the characteristic features of these two diseases in asthmatic patients are briefly described. Concerning the association, the authors have found that some authors suggest a common nosological framework with many transitional forms.     

Case of bronchocentric granulomatosis that became recognized in the course of allergic bronchopulmonary aspergillosis]

A 56-year-old man with allergic bronchopulmonary aspergillosis (ABPA) was admitted due to the appearance of nodular opacities in the right upper lung field on chest radiography, after discontinuing itraconazole and clarithromycin on the suspicion of possible hepatic adverse effects. Chest CT scans on admission revealed nodular opacities in the right S3 and lingula bronchus, and bilateral bronchiectasis with mucoid impactions. A specimen obtained by transbronchial lung biopsy showed complete replacement of bronchioles by necrotizing granulomatous inflammation, containing the diagnosis of bronchocentric granulomatosis. Treatment with corticosteroids and micafungin sodium resulted in marked resolution of nodular opacities and mucoid impacts. This case suggests that abrupt cessation of antifungal agents and macrolides may provoke acute exacerbation of ABPA and development of bronchocentric granulomatosis.     

Pulmonary giant bulla in Wegener's granulomatosis

The association of lung emphysema with severe systemic antineutrophil cytoplasm antibodies (ANCA)-positive vasculitis, such as Wegener's granulomatosis is unusual since only four cases have been described previously. We report the first case of a 30 year-old smoker man presenting with biopsy-proven Wegener's granulomatosis, who developed a bullous emphysema during severe active lung vasculitis, in association with positive ANCA disclosing an anti-myeloperoxydase pattern. Alpha 1-antitrypsin deficiency, a known risk factor of lung emphysema recently found to be associated with anti-proteinase 3-positive vasculitis, was not present in this patient. Cigarette smoking, in association with severe lung vasculitis, might have contributed to the development of this emphysematous lesion.     

Pulmonary tuberculosis

Pulmonary tuberculosis is a major cause of morbidity and mortality worldwide, resulting in the greatest number of deaths due to any one single infectious agent. This trend is due, at least in part, to increasing numbers of individuals co-infected with HIV and Mycobacterium tuberculosis (MTB). Concerted efforts between the World Health Organization and other agencies, therefore, are underway to improve tuberculosis control worldwide. These include basic research in tuberculosis diagnostics and vaccine development, institution of preventive therapy in individuals dually infected with HIV and MTB, and directly observed short-course antituberculous therapy in developing countries with a high prevalence of MTB infection. Further, newer, longer-acting antituberculous therapeutic agents such as rifapentine, which allow twice-weekly dosing in the continuation phase of anti-MTB therapy, have recently been released and are undergoing clinical trials. This review provides a synopsis of recent developments in these areas and serves as a reference source for interested readers.     

A case of bronchocentric granulomatosis associated with Aspergillus that was difficult to differentiate from lung cancer]

We encountered a case of suspected bronchocentric granulomatosis (BCG) occurring in a COPD patient. Chest CT on admission showed a nodular shadow with indentation and a notch-like opacity in right S(3)a. Based on these findings, lung cancer was suspected. As we could not establish a definitive diagnosis, video-assisted thoracic surgery was performed. The histological findings of the resected specimen demonstrated BCG with clustering of fungal hyphae in the bronchioles. As the lesion was resected completely, we did not give additional treatment with an antifungal agent. It is difficult to differentiate BCG from lung cancer preoperatively. However, considering the homogeniecity of the nodule, it may be able to differentiate this type of lesion from lung cancer based on the feature. We report a rare case of a solitary nodule caused by BCG with Aspergillosis.     

Pulmonary immune complex deposition in Wegener's granulomatosis

Two male patients with pulmonary manifestations of Wegener's granulomatosis are presented. One had an elevated rheumatoid factor, and both had elevated levels of immunoglobulin E. Both demonstrated characteristic necrotizing granulomatous lesions on light microscopy of lung tissue. Immunohistologic analysis of lung tissue demonstrated a granular deposition of immunoglobulin G and complement. Raji cell assay of sera demonstrated elevated levels of circulating immune complexes in the sera of the one patient tested prior to any therapy. These findings support the hypothesis that immune complex deposition contributes to the pathogenesis of Wegener's granulomatosis.     

韦格纳肉芽肿病肺损害的临床分析

目的 了解韦格纳肉芽肿病(wegener’s granulomatosis)肺损害的情况。方法 回顾性分析近20年来明确诊断的20例，韦格纳肉芽肿病患的临床资料。结果 (1)12例(60％)有肺损害，症状为咳嗽、脓痰、血痰、咯血、呼吸困难等，其中以肺部症状为首发表现5例。(2)肺部影像学表现为：肺部多发性病变，可单侧、双侧，以双侧为多(8／12)；病变多样，可呈大小不等的结节状影，并可形成厚壁或薄壁空洞，亦可呈渗出性斑片状改变，易被误诊为肺脓肿(本组4例)、肺结核(本组3例)、肺炎(本组5例)等。经治疗后，病灶可明显吸收或消失。(3)明确病理诊断的患中，多部位、多次活检10例，肺多次活检4例。结论 韦格纳肉芽肿病常见肺损害，肺影像学表现为多发、多形态病灶，易被误诊为肺脓肿、肺结核、肺炎、肺癌等，应注意鉴别。多部位、多次肺活检有助于明确诊断。     

Pulmonary talc granulomatosis in a cocaine sniffer

The development of pulmonary granulomatosis following intravenous injection of medications intended for oral use has been well described previously. Talc is the most commonly implicated agent. We present a case of talc granulomatosis which developed in a patient following cocaine sniffing and suggest that this may be the cause of development of granulomata in drug addicts who deny any history of intravenous drug abuse.     

Pulmonary and osteoarticular tuberculosis

Tuberculosis is an important illness with an increasing prevalence in the last years in part due to AIDS. Considered primarily a pulmonary disease, tuberculosis can also affect almost any body system (nervous, genitourinary, and gastrointestinal as well as bones and joints) through lymphohematogenous spread. The authors present a case of pulmonary and osteoarticular tuberculosis diagnosed simultaneously.     

Pulmonary lymphomatoid granulomatosis radiologically mimicking interstitial pneumonia]

We report a rare case of pulmonary lymphomatoid granulomatosis radiologically mimicking interstitial pneumonia. A 57-year-old man was admitted to our hospital because of chest bilateral reticular shadow with sustained cough and breathlessness for 10 years. Chest CT scans showed multiple ground-glass opacities, traction bronchiectasis and cystic change in both lungs, in addition to hilar and mediastinal lymphadenopathy. A histopathologically diagnosis of pulmonary lymphomatoid granulomatosis (angiocentric immunoproliferative lesion, grade 1) was made by thoracoscopic lung biopsy. In this case, serological and immunohistochemical analyses did not show Epstein-Barr virus infection. No clinical or radiological deterioration has been observed thereafter despite no medication.