Inflammatory pseudotumor of the lung]

The patient was a 76-year-old man whose chief complaint was a dry cough. His chest X-ray film revealed a large hazy shadow with unclear margin in the left upper lobe. Bronchiolitis obliterans organizing pneumonia was initially diagnosed because transbronchial lung biopsy (TBLB) specimens showed organizing pneumonia with no evidence of malignancy. However, because the hazy shadow increased gradually in size despite steroid therapy. TBLBs were performed several more times to confirm the diagnosis. The last TBLB specimen showed proliferation of fibroblasts and mononuclear cells, with marked infiltration mainly of plasma cells 12 months after the initiation of steroid therapy. Because we were unable to obtain a histological diagnosis by bronchofiberscopy, a left upper lobectomy was preformed and the lesion resected. Histology disclosed inflammatory pseudotumor of a lymphoplasmacytic type with organizing pneumonia. The results of an immuno-histochemical examination confirmed that the proliferating plasma cells were polyclonal. These findings suggest that inflammatory pseudotumors should be taken into account by differential diagnoses of cases of organizing pneumonia that are resistant to steroid therapy.     

Two cases of inflammatory pseudotumor of the lung

Two cases of inflammatory pseudotumor of the lung were reported. Case 1. A 68 year-old man was referred to our hospital because of an abnormal shadow on chest X-ray. He had a history of pneumonia in the right upper lobe five months before. The chest X-ray film revealed a coin lesion in the right upper lung field (S1), the same segment as the previous pneumonia. Although RPF and INH were administered for three months, the shadow did not change, and cough and sputum continued. In order to confirm the diagnosis, open thoracotomy was performed and microscopic findings of the resected tumor showed inflammatory pseudotumor; proliferation of fibrous tissue with infiltration by inflammatory cells (plasma cells, lymphocytes and a few neutrophils). Case 2. A 35 year-old man was admitted to our hospital because of an abnormal shadow on chest X-ray, i.e. a coin lesion with vascular indentation in the left lower lung field (S8). A wedge resection including the mass was performed, and histopathologic examination revealed inflammatory pseudotumor, or plasma cell granuloma; proliferation of fibrous connective tissue with infiltration by predominantly mature plasma cells. Both patients have been doing well after the operation. We also reviewed 46 cases reported in the Japanese literature and discussed various aspects of this disease. Of 10 patients who had a history of previous respiratory tract infection, 8 had histopathologic features with various inflammatory cells, including many lymphocytes, as in our Case 1. Although the common etiology of inflammatory pseudotumor is obscure, we suppose that in some cases the lesion may be a result of post-inflammatory repair process.     

Wegener's granulomatosis in a woman with asymptomatic primary biliary cirrhosis

A 60-year-old woman was admitted to our hospital in June 1985, complaining of fever, cough and right lower chest pain, with a five-year history of asymptomatic primary biliary cirrhosis. Chest X-ray on admission showed an infiltrative shadow in the right lower lung field. She was first treated with various antibiotics unsuccessfully. Hemoptysis continued. Dyspnea and anemia appeared. Chest X-ray 17 days after admission showed multiple infiltrative shadows in the both lung fields. She was treated with steroid pulse therapy successfully. During prednisolone treatment decreasing nodular shadows with cavities appeared on chest X-ray. An open lung biopsy was performed in March 1986. The histologic findings showed a necrotizing vasculitis with granuloma and perivascular fibrosis. She was treated with prednisolone and prednisolone-azathioprine therapy unsuccessfully, but successfully with prednisolone-cyclophosphamide therapy. This case was a rare case of Wegener's granulomatosis with transition from fulminant type to granulomatous type. No similar case of Wegener's granulomatosis with asymptomatic primary biliary cirrhosis has been reported in the literature.     

A case of idiopathic pulmonary fibrosis with chronic eosinophilic pneumonia

A 58 year-old man was admitted to our hospital with complaints of dyspnea and cough. Chest X-ray examination revealed diffuse nodular shadow, infiltration in the left upper and lower lobes, and volume loss. BALF showed increased number of cells, particularly eosinophils (65.6%). Eosinophilia (17-13%) was also seen. The histological examination of the TBLB specimen revealed irregular thickening and edema of alveolar septa. From these findings together with clinical features the case was diagnosed as eosinophilic pneumonia. Oral administration of prednisolone started with 30 mg. Within a week, eosinophilia in peripheral blood decreased to 1%, PaO2 increased to 87.5 Torr in room air, and CRP became negative. His chest X-ray film showed the disappearance of infiltrative shadows in the left upper and lower lobes. On the 19th day was prednisolone decreased to 20 mg. Laboratory data and chest roentgenogram showed exacerbation, and PaO2 gradually decreased to 62.9 Torr. Patchy shadows were seen in the right upper lobe. The dose of prednisolone was again increased to 60 mg/day, and was quite effective. The drug dosage was tapered by 10 mg/10 days to 40 mg/day, however all data exacerbated again. The clinical course seemed peculiar for eosinophilic pneumonia, however small reticulonodular shadows on chest X-ray did not change. Therefore, open lung biopsy was performed. The histology of the specimen showed fibrosis, lymphocyte infiltration and thickening of alveolar septa. The clinical course and histopathological findings led to a diagnosis of idiopathic pulmonary fibrosis accompanied with chronic eosinophilic pneumonia.     

Inflammatory pseudotumor of the lung with infiltration identified radiographically and hemoptysis]

The patient was a 53-year-old man whose chief complaint was a dry cough and hemoptysis. Chest X-ray films and computed tomographic scans revealed a hazy shadow with unclear margins in the left middle lobe. A diagnosis of inflammatory pseudotumor was made because transbronchial lung biopsy specimens showed proliferation of plasma cells and lymphocytes. Initially, in response to steroid therapy, his clinical condition and pulmonary infiltration improved, but about 30 days after the start of steroid therapy, hemoptysis occurred and the shadow increased, and therefore left segmentectomy was performed. Histology revealed not only proliferation of acute and chronic inflammatory cells but also changes in the vessels. These pathologic findings were considered to be related to the infiltration shadows and the hemoptysis.     

An adult case of cervico-mediastinal lymph nodes tuberculosis followed by the development of pulmonary lesions during the treatment with antituberculous drugs

A 27-year-old man was admitted to our hospital due to a painful mass in the right neck and fever. Cervical and superior mediastinal computed tomography showed an enlargement of right supraclavicular lymph node and multiple swollen mediastinal lymph nodes, including low-density areas and contrast medium-enhanced septa and margins. Smears of the pus obtained from right supraclavicular lymph node showed acidfast bacilli identified as Mycobacterium tuberculosis by PCR method. He was treated with antituberculous drugs with INH, RFP, EB, and PZA. PZA was given for initial two months. Six months later, productive cough developed and chest X-ray films showed infiltrative shadow in the right upper lung field. One month after the onset of cough, bronchoscopy revealed a polypoid lesion with a white coating in the right main bronchus. Microscopic examination of the specimen obtained by transbrochial biopsy revealed many epithelioid cell granulomas, consistent with tuberculosis. From these findings, pulmonary lesion was suggested to be due to invasion of the mediastinal lymph node into the bronchus. After one year of antituberculous chemotherapy, the swelling of the cervical-mediastinal lymph nodes was reduced and the abnormal chest X-ray shadows disappeared.     

A case of pericostal abscess exacerbated during antituberculous chemotherapy

A 27 Year-old female with pulmonary tuberculosis in right upper lobe developed right pericostal abscess during the course of antituberculous chemotherapy. The chest x-ray films on first admission showed infiltration with cavity formation and nodular shadows in the right upper lung field. Seven months after starting the antituberculous therapy with INH, RFP and EB, a new tumorous shadow appeared in the right chest wall. Microscopic examination of the specimen obtained by needle aspiration biopsy disclosed positive acid-fast bacilli. Because of the ineffectiveness of drug therapy on lesions in the right chest wall, surgical treatment was performed and the disease was diagnosed as pericostal abscess.     

Bronchioloalveolar carcinoma complicated by a lesion resembling bronchiolitis obliterans organizing pneumonia in the opposite lung]

A 50-year-old woman with abnormal shadows in her chest radiograph was admitted for a more detailed examination in April 2000. A chest CT scan showed infiltrates surrounded by ground-glass opacities in the right upper lobe, and ground-glass opacities with partial infiltrative changes in the left lower lobe. A diagnosis of adenocarcinoma like bronchioloalveolar carcinoma (BAC) was made for the lesion in the right upper lobe by means of a transbronchial biopsy. However, the specimen from the left lower lobe showed nonspecific changes, and bronchiolitis obliterans organizing pneumonia (BOOP) or a metastatic lesion from the BAC was suspected. We prescribed steroids for the short term. Since the lesion in the left lobe disappeared, we performed a right upper lobectomy. In cases of primary lung carcinoma, BOOP in the opposite lung is rare and it was difficult to decide on the operability. We concluded that steroid therapy is useful for distinguishing between inflammatory and tumorous lesions in cases in which a detailed examination is difficult.     

A case of pulmonary eosinophilic granuloma with involvement of the ocular fundus]

A 37-year-old man was admitted to our hospital because of abnormal shadows on chest X-ray film at routine medical examination. The chest X-ray showed diffuse granular shadows in the bilateral upper and middle lung fields. Chest CT showed multiple cystic lesions and nodular lesions. Bronchoalveolar lavage (BAL) and transbronchial lung biopsy (TBLB) were performed. Electron microscopic examination of BAL fluid revealed Langerhans' cells, and lung tissue specimen obtained by TBLB showed granuloma with infiltration of histiocytes and eosinophils. During the clinical course, the patient complained of cough and decrease of right visual acuity in association with an increase of abnormal shadows on chest X-ray. The lung disease gradually resolved after the cessation of smoking in spite of no therapy. In the right eye, an exudative lesion with retinal elevation was noted in the ocular fundus. Corticosteroid administration was effective for the treatment of this lesion. There was no pathological evidence of the right ocular lesion. However, the correlation of its clinical course with the lung disease suggests a relationship between the ocular lesion and pulmonary eosinophilic granuloma.     

A case of migratory air space infiltration after radiation therapy for breast cancer]

A 54-year-old woman underwent conserving surgery for right breast cancer, and received a cumulative dose of 50 Gy of radiation therapy to the remaining part of the right breast. About five months after the termination of irradiation, cough and low-grade fever developed. The chest radiograph showed an infiltrative shadow in the right lung field. Organizing pneumonia was identified in the transbronchial lung biopsy specimen. After prednisolone was given to the patient the clinical symptoms and infiltrates seen in the radiograph disappeared. In the course of tapering the prednisolone dose, new infiltrative shadows developed in the upper right lung and the left lung. The histologic changes were shown by transbronchial lung biopsy to be organizing pneumonia. The increased dose of prednisolone resulted in the rapid improvement of the clinical symptoms and chest radiograph abnormalities. This case suggests that breast radiation after conserving surgery for breast cancer may cause a pathologic process similar to that of bronchiolitis obliterans organizing pneumonia.     

A case of subacute necrotizing lymphadenitis with localized interstitial pneumonia

A 16-year-old male was admitted with a history of cervical lymph node swelling, high fever, cough and hemosputum. On admission, bilateral cervical lymph nodes swelling and fine crackles in the right lower lung field were noted. A chest X-ray film showed an infiltrative shadow in the right lower lung field and right hilar enlargement. Cervical lymph node biopsy specimens revealed wide areas of necrosis with nuclear debris. Transbronchial lung biopsy showed infiltration of lymphocytes in the interstitium and bronchoalveolar lavage fluid showed increased T-lymphocytes and a decreased T4/T8 ratio. The patient was diagnosed to have subacute necrotizing lymphadenitis with T-lymphocyte alveolitis. Additionally, antinuclear antibodies were positive, and anti HTLV-I antibody was false positive. These findings suggested an immunological abnormality in this case. His cervical lymph node swelling and infiltrative shadow on chest X-ray film improved with steroid therapy. Our case may be the first case of subacute necrotizing lymphadenitis with T-lymphocyte alveolitis.     

A case of Beh?et's disease with hemoptysis and chest X-ray abnormality

A 41-year-old man was admitted to our hospital because of hemoptysis and dyspnea. Physical examination disclosed oral ulcer, acne and scrotal ulcer. Although ocular lesions were absent, the needle reaction was positive. Then we diagnosed this case as incomplete type of Beh?et's disease. Chest X-ray showed diffuse retinolinear shadows in the entire lung field and an infiltrative shadow in the left middle lung field. CT-scan revealed interstitial shadows in the entire lung field, a wedge-shaped shadow in left S6 and aneurysmic dilatation of the left lower lobe pulmonary artery. Pulmonary angiography showed obstruction and aneurysmic dilatation of the left lower lobe pulmonary artery. Pathological examination of the TBLB specimen revealed thickening of the vessel wall, perivascular infiltrations of mononuclear cells, hemorrhage and slight thickening of alveolar septae. These findings were compatible with pulmonary lesions of Beh?et's disease. After administration of corticosteroid, the symptoms and the chest roentgenographic findings improved. Pulmonary lesions of Beh?et's disease were discussed.     

A case of asymptomatic paraganglioma diagnosed by abnormal shadow on chest X-ray]

A 65-year-old man was noted to have an abnormal shadow on chest X-ray at mass screening examination, and was seen at Douhoku Byoin National Sanatorium. Chest X-ray showed a homogeneous mass shadow and a lytic lesion of the right 5th rib with extra-pleural tumor sign at the right upper and middle lung fields. Retroperitoneal tumor of 5 cm in diameter was detected in the right adrenal area by ultrasonography. Because of increasing plasma levels of norepinephrine and dopamine, the diagnosis of malignant pheochromocytoma with rib metastasis was made. Histologically, chromogranin staining was positive in each resected specimen. The retroperitoneal tumor was separate from the right adrenal gland. We report a rare case of malignant paraganglioma which was asymptomatic in spite of increasing plasma levels of catecholamines.     

Case of primary cavitary sarcoidosis: exacerbation after 16 years from onset]

A 46-year-old man presented with the complaint of cough, fever and dyspnea on exercise. Sixteen years previously stage II sarcoidosis was diagnosed, with an ocular lesion. So far, he had had no respiratory symptoms and was followed up without medication. Two months before his visit, he had a cough. He gradually developed a fever and felt dyspnea on exercise. Chest X-ray showed an infiltrative shadow with a cavity in the right upper lobe. Bronchofiberscopy was performed and the biopsy specimen from the cavity wall revealed noncaseating epitheloid cell granulomas, suggesting primary cavitary sarcoidosis. Corticosterid therapy was started. His symptoms improved rapidly, and the infiltrative shadow on the right upper lobe decreased within one month. The cavity wall was thin on the following computed tomography. Thirty-two cases of primary cavity sarcoidosis were reported in Japan from 1975-2005. The mean age of the patients was 26.5 years old and the ratio of males to females was 13:3. In most cases, corticosteroid therapy was effective and nine spontaneously improved cases were reported. To determine the indications for steroid therapy, further examination is needed.     

A case of Sweet's syndrome with a variety of chest radiological findings]

We report a rare case of Sweet's syndrome (acute febrile neutrophilic dermatosis) with a variety of chest radiological findings. A 73-year-old man, who had been treated with corticosteroid for Sweet's syndrome for 2 years, was admitted to our hospital because of pyrexia with a infiltrative chest shadow. Chest CT scans showed consolidation and ground-glass opacities with air-bronchogram in the right lower lobe. Treatment with antibiotics seemed to be effective but there was no improvement of chest shadows. Simultaneously with his pyrexia, diffuse centrilobular-micronodular shadows and a mass-like shadow appeared on chest CT after 2 months and after the next 2 months, respectively. Bronchoalveolar lavage fluid contained increased neutrophils but not any infectious pathogen. Transbronchial lung biopsy specimens revealed chronic interstitial infiltrate with alveolar wall thickening and neutrophil accumulation in the airspace. A diagnosis of pulmonary involvement in a patient with Sweet's syndrome was finally made and he was successfully treated with corticosteroid.     

A case of carcinosarcoma of the lung]

A 78-year-old man was admitted to our hospital with cough and left anterior chest pain. Chest X-ray examination on admission revealed a tumor shadow in the left upper lobe. Malignant tumor cells were observed on histopathological examination of a specimen obtained by bronchoscopic biopsy. Radiotherapy was performed but was not effective, and the patient died of respiratory failure 4 months after admission. Autopsy revealed a 15 cm diameter tumor with marked local invasion tendency without distant metastasis. Microscopically, the tumor consisted partly of squamous cell carcinomas, and partly of fibrosarcomas, composed of spindle cells and osteo-chondrosarcoma. The tumor was therefore diagnosed as carcinosarcoma. Immunohistochemical examination showed positive keratin and EMA staining only in the squamous cell carcinoma component of the tumor.     

Case of pulmonary Mycobacterium mageritense infection: the difficulty of differential diagnosis of granulomatous lung diseases

A 36 year-old female was pointed out of pulmonary abnormal shadows in the annual chest survey. Chest radiograph and computed tomography (CT) disclosed bilateral diffuse infiltrative shadows and tree-in-bud appearance in the right upper lung field and the left lingula. A sputum smear for acid-fast bacilli was negative. Histopathologically, the transbronchial lung biopsy specimen revealed non-caseous epithelioid granulomas with numerous giant cells. Acid-fast bacilli were cultured from her sputum, however, nontuberculous mycobacteria was not detected by DNA-DNA hybridization method. Mycobacterium mageritense was identified by 16S ribosomal RNA sequencing with 100% matching. The isolated colony of M. mageritense was resistant to nine anti-tuberculous drugs. Follow-up chest CT scan showed a gradual decrease of infiltrative shadows without therapy. To the best of our knowledge, M. mageritense infections are rare, and this is the first case report of pulmonary infection in the literature. We conclude that the pulmonary infection of M. mageritense is one of causes of granuloma formation, and in some case it is difficult to differentiate clinically from sarcoidosis.     

A case of smoldering adult T-cell leukemia complicated by various pulmonary infections]

A 46-year-old man was admitted to Oita Medical College Hospital on October 16, 1987, because of cough and sputum. Chest X-ray and chest CT films showed diffuse reticulonodular shadow. The specimens obtained by transbronchial lung biopsy revealed cysts of pneumocystis carinii. Abnormal lymphocytes with lobulated nuclei were found 2-7% of peripheral leucocytes. The anti HTLV-I antibody was positive. According to these data, we diagnosed the patient as smoldering adult T cell leukemia with pneumocystis carinii pneumonia. The abnormal shadow on chest X-ray disappeared after SMX-TMP and pentamidine treatment. After about 1 year, he was again admitted for high fever. Chest X-ray showed infiltration with cavity in right upper lobe. Streptococcus pneumoniae was isolated from the sputum. The infiltration shadow on chest X-ray disappeared after antibiotics treatment. However, multiple nodular shadow appeared on the chest X-ray and ATL cell infiltration was found in the specimens of transbronchial lung biopsy. ATL cells in peripheral blood also increased and serum LDH and Calcium levels were markedly high. According to these data, we diagnosed the patient as having a ATL crisis. Although chemotherapy for ATL was started, the ATL, cell infiltration shadow on the chest X-ray enlarged, and bilateral diffuse patchy shadows was appeared on the chest X-ray. He died of respiratory failure on April 26, 1989. Cytomegalovirus pneumonia and ATL cell infiltration were revealed by necropsy.     

A case of cervical-mediastinal lymph node tuberculosis progressed to pulmonary lesion through a bronchial fistula]

The patient was a 25-year-old man who had been admitted to a local hospital due to fever and trachelophyma. Tubercle bacillus was detected in pus culture obtained by biopsy of the trachelophyma, but not in sputum culture. Because combined therapy with 3 antituberculous drugs (RFP, INH and SM) failed to reduce the fever or drainage from the biopsy region, the patient was transferred to our hospital. Chest X-ray films taken on admission revealed dilatation of the superior mediastinal shadow; chest CT images revealed cervical and mediastinal lymphadenopathy and an anterior mediastinal abscess, but no pulmonary lesion. About 2 months after admission, cough developed and Gaffky type 2 was detected in the patients sputum. Bronchoscopy and bronchography revealed a bronchomediastinal fistula. Forty days after the onset of cough, reticulogranular shadows were observed in the right upper lobe on chest X-ray films, and a diffuse centrilobular lesion was observed in the right upper lobe on chest CT images. From these clinical observations, the patient was given a diagnosis of cervical-mediastinal lymph node tuberculosis, which had progressed to pulmonary lesion through a bronchial fistula due to lymphadenitis.     

A case of severe adult measles pneumonia--efficacy of combination of steroid pulse therapy, high-dose vitamin A and gamma globulins]

A 33 year-old female was admitted with facial, trunk and limb eruptions, conjunctiva intrahemorrhage, Koplik's spots in the pharynx and severe hypoxemia after fever and upper respiratory tract symptom. Infiltrative shadow of the whole right lung was seen on chest radiography. Fine crackles were seen in the lower left lung and in the whole right lung. Severe inflammation and liver dysfunction were indicated by blood test. Measles antibody IgM was high. The abnormal interstitial shadows were confirmed in greater detail by chest computed tomography. Her condition was diagnosed as measles pneumonia. A combination therapy with steroid pulse, high dose vitamin A, and gamma globulin was started, after which the patient gradually improved, indicating the effectiveness of this combination therapy for severe adult measles pneumonia.