A case of chronic necrotizing pulmonary aspergillosis]

A 70-year-old woman with diabetes mellitus who was following a therapeutic diet showed an infiltrative shadow in the right upper lung field on chest roentgenogram in April, 1986. She was diagnosed as having pneumonia and was treated for five months with several antibiotics, but the abnormal shadow on chest roentgenograms increased in size. Therefore, she was admitted to our hospital in October 1986. Although tubercle bacilli were not isolated from her sputum or from materials obtained by bronchoscopic examination, we made an initial diagnosis of pulmonary tuberculosis based on the findings of chest roentgenograms, tomographs and CT scanning. In spite of treatment with antituberculous drugs, the infiltrative shadow with cavity on chest roentgenograms continued to increase in size, and the patient developed occasional hemoptysis. Percutaneous needle biopsy was performed in February 1987 to establish a definite diagnosis, and the presence of Aspergillus fumigatus was confirmed by microscopic examination and culture. After treatment with miconazole and 5-FC for 3 to 4 months, the abnormal shadow on the chest roentgenogram gradually disappeared and was almost undetectable one year later. The clinical course of this patient was considered to be strongly indicative of chronic necrotizing pulmonary aspergillosis, which was described by Binder et al. in 1982.     

A case of hyper-IgE syndrome complicated by chronic necrotizing pulmonary aspergillosis]

A 46-year-old man afflicted with recurring infection and bone fracture consulted our hospital because of general malaise and increase of sputum. He was given a diagnosis of chronic necrotizing pulmonary aspergillosis, and underwent right lower lobectomy. Six months later, chronic necrotizing pulmonary aspergillosis become exacerbated. Slightly improvement was obtained with voriconazole. Two months later, this disease become reactivated, and slightly improved with itraconazole and amphotericin B. Subsequently, hyper-IgE syndrome was diagnosed in him by pathognomonic face, recurring infection and bone fracture, chronic necrotizing pulmonary aspergillosis, elevated IgE, and eosinophilia. We suggested that the pathogenic cause of chronic necrotizing pulmonary aspergillosis in this case was hyper-IgE syndrome. After that, chronic necrotizing pulmonary aspergillosis was reexacerbated. We added micafungin, increased itraconazole, interferon gamma, and so on. As a result, his chest radiograph and symptoms improved slowly. Cases of hyper-IgE syndrome are rare.     

A case of chronic thromboembolic pulmonary hypertension based on essential thrombocythemia]

We report a case of chronic thromboembolic pulmonary hypertension based on essential thrombocythemia. A 72-year-old woman was admitted to our hospital with dyspnea. The hematologic workup revealed a platelet count of 99.2 x 10(4)/microliter. Chest radiographic examination revealed cardiomegaly with bilateral pulmonary artery enlargement. A perfusion lung scan suggested and pulmonary angiography confirmed--multiple pulmonary embolism. Pulmonary artery pressure was 90/30 (51) mmHg. Thrombolytic therapy was performed successfully, and a diagnosis of essential thrombocythemia was made on the basis of the criteria proposed by the Polycythemia Vera Study Group. The therapy of essential thrombocythemia including ranimustine was effective, and one year later, the essential thrombocythemia and chronic respiratory failure had improved. To our knowledge, this case of chronic thromboembolic pulmonary hypertension based on essential thrombocythemia is a very rare one.     

A case of pulmonary coccidioidomycosis]

A 32-year-old Japanese woman who had been living in Arizona, United States since two years previously was admitted to our hospital because of an abnormal shadow. Her chest X-ray films revealed a small cavity in the right middle lung field which had not been identified two years ago. The chest CT showed that it was 15 mm in diameter and located in the right S6, and open lung biopsy was performed. Histological examination revealed caseous lesion of the epithelioid granulomas, that contained spherulitic forms of endospores. Fungal cultures demonstrated barrel-shaped arthroconidia. A diagnosis of pulmonary coccidioidomycosis was made.     

A case of pulmonary leiomyosarcoma]

A 73-year-old woman with hemoptysis visited our hospital. Chest radiography showed a massive shadow on the right middle lung field. Bronchofiberscopic examination demonstrated a polypoid tumor obstructing the right middle lobe bronchus. A chest CT scan showed that the tumor was situated in the right middle lobe, obstructing the right pulmonary artery trunk. Sarcoma was diagnosed after a CT-guided biopsy. The tumor grew rapidly, giving rise to brain metastasis, which led to the death of the patient. An autopsy examination confirmed the diagnosis as pulmonary leiomyosarcoma.     

A case of chronic necrotizing pulmonary aspergillosis due to Aspergillus flavipes associated with pulmonary fibrosis]

The patient was a 76-year-old man with complaints of fever, productive cough, and purulent sputum. He was on home oxygen therapy for pulmonary fibrosis. Antibiotics were administered, but with no improvement. Because the inflammatory condition persisted, the patient was admitted. Computed tomographic (CT) scans of the chest demonstrated infiltrative shadows and a fungus ball, and pulmonary aspergillosis was suspected. The patient was given an antimycotic agent and his symptoms gradually resolved. Subsequent chest CT scans revealed a reduction of the infiltrative shadows, but no reduction in the size of the fungus ball. Aspergillus flavipes was cultured from samples of bronchoalveolar lavage fluid. Aspergillus fumigatus is the most common causative agent of pulmonary aspergillosis. In this respect, our case was considered rare and of value to the diagnosis and treatment of future cases of pulmonary aspergillosis.     

A case of chronic necrotizing pulmonary aspergillosis diagnosed using percutaneous intracavitary endoscopy]

A 65-year-old man with pneumoconiosis visited our hospital for dyspnea on effort. Chest radiography and computed tomography on admission showed cavities with an air-fluid level, consolidation in the right lower lung, and right pleural effusion. The thoracic cavity and an infected cyst were drained, and antibiotics were administered. On detection of molds like Aspergillus species and of aspergillus antigen from the sputum, aspergillosis was suspected and amphotericin B was administered intravenously. Renal dysfunction caused by amphotericin B led to its withdrawal. Abnormal shadows in the chest radiographs and computed tomograms did not improve. To aid in diagnosis, percutaneous intracavitary endoscopy was performed. Yellow-white mural nodules resembling cauliflower were found on endoscopic examination, and a biopsy specimen of the nodules showed hyphae of Aspergillus. Aspergillus fumigatus was cultured from the intracavity fluid. Pulmonary aspergillosis was diagnosed and amphotericin B was administered via the drainage catheter in order to protect renal function. The abnormal shadows then disappeared and the subsequent clinical course was good. In this case, percutaneous intracavitary endoscopy was useful in diagnosing pulmonary aspergillosis.     

A case report of pulmonary blastoma--biphasic pulmonary blastoma]

We report a biphasic pulmonary blastoma in a 77-year-old man. In a routine chest radiographic examination, in the left upper lung field, a massive shadow that had been entirely absent one year before was detected. The preoperative imaging films showed a 10 x 8 cm, well-circumscribed solid tumor in the upper lobe of the left lung. A preoperative clinical diagnosis of primary lung cancer was considered. The intraoperative findings were that the tumor had invaded the visceral pleura with adhesion to the parietal pleura. Left upper lobectomy with lymph node sampling was performed, and since invasion of the pulmonary artery wall was confirmed, complete removal of tumor was not possible. The postoperative diagnosis was biphasic pulmonary blastoma. The patient was treated with radiotherapy up to a limit of 50 Gy, covering the area around of the left hilum. At the latest follow-up, 1 year postoperatively, the patient was clinically and radiologically free of the disease.     

A case of loxoprofen-induced pulmonary eosinophilia]

A 65-year-old female suffering from lumbago, headache, and hypertension had been treated with nonsteroidal anti-inflammatory drugs (NSAIDs) and antihypertensive drugs. On June 13, 1990, 2 weeks after the commencement of loxoprofen administration, she developed cough and low grade fever. She was treated with antibiotics and NSAIDs without improvement. Laboratory data showed marked eosinophilia (2200/mm3), elevation of IgE (3090 IU/ml), and liver dysfunction. Her chest X-ray revealed no active lesion, but the percentage of eosinophils in BALF was elevated (38%). Because drug-induced eosinophilic pneumonia was suspected, all drugs were discontinued. Her symptoms improved and the abnormalities of laboratory data normalized. The lymphocyte stimulation test was weakly positive with three NSAIDs (loxoprofen, pranoprofen, and alminoprofen). The challenge test by loxoprofen reproduced eosinophilia and liver dysfunction, suggesting that she had loxoprofen-induced eosinophilic pneumonia. To our knowledge, this is the first reported case of loxoprofen-induced lung injury.     

A case of multidrug-resistant pulmonary tuberculosis]

A 44-year-old woman with multidrug-resistant pulmonary tuberculosis was admitted to our hospital in August 1998. She had been treated with the anti-tuberculosis agents isoniazid (INH), rifampicin (RFP), pyrazinamide and streptomycin (SM) for two months. However, tubercule bacilli found in a sputum culture on admission showed resistance to INH, RFP and SM, and so these agents were replaced with kanamycin (KM), ethionamide, cycloserine and levofloxacin. Unfortunately, the bacilli persisted in the sputum smears, and the patient complained of prolonged pain in the sites of intramuscular injection of KM. In January 1999, inhalation of KM was begun, resulting in the disappearance of the bacilli from the sputum and in improvements in chest radiographs. Inhalation of KM could be an effective therapy, with fewer adverse effects, in cases of multidrug-resistant pulmonary tuberculosis.     

A case of true pulmonary carcinosarcoma]

A 72-year-old male was admitted to our hospital because of dyspnea. A chest radiograph showed a mass shadow measuring 40 x 40 mm in the left lower lobe, and fiberoptic bronchoscopy revealed obstruction of the left B8 by the tumor. Adenocarcinoma was suspected after a transbronchial biopsy, and left lower lobectomy was performed. Histologically, the resected specimen was composed of carcinomatous and sarcomatous elements, and further immunohistological study revealed that the former component was poorly differentiated squamous cell carcinoma and the latter was fibrosarcoma. We therefore concluded that the tumor was a true carcinosarcoma of the lung.     

A case of pulmonary arteriovenous fistula]

A 31-year-old man presenting with abrupt onset of convulsions was transported by ambulance to a nearby neurosurgery hospital. Imaging examination of the brain revealed a brain abscess in the left parietal lobe. Surgical excision of the abscess was performed. After the operation, the patient was referred to our hospital because of abnormal radio-opacities on the chest X-ray film, which were subsequently diagnosed to represent pulmonary arteriovenous fistulae in the lower lobes of both lungs. The fistulae were successfully embolized with detachable coils. Orthodeoxia (worsening of hypoxemia on assuming the upright from the supine position) was confirmed by measuring the partial pressure of oxygen in the arterial blood. We examined the shunt ratio by both the 100% O2 inhalation and 99mTc-MAA methods, and a higher ratio was obtained with the radioisotope method than with the 100% O2 inhalation method. Although it is known that the 99mTc-MAA method is more precise than the 100% O2 inhalation method, the latter is easier to perform and therefore more useful in practical clinical situations.     

A case of pulmonary capillary hemangiomatosis]

In March 1995, a 54-year-old woman was admitted to a hospital because of exertional dyspnea and her condition was diagnosed as primary pulmonary hypertension. In September 1996, respiratory difficulty and hemosputum were woted. The chest computed tomographic scan showed diffuse micronodular opacities, and she was admitted to our hospital for examination of other pulmonary conditions giving rise to pulmonary hypertention. However no lung biopsy could be performed, because the pulmonary arterial pressure was too high. Although the patient received medication, she died of progressive right heart failure. The pathological diagnosis at autopsy was pulmonary capillary hemangiomatosis (PCH). Although PCH is a very rare disease that causes secondary pulmonary hypertension, it is important to distinguish it from primary pulmonary hypertension and pulmonary veno-occlusive disease.     

A case of pulmonary parenchymal endometriosis]

In 1982, a 45-year-old woman was hospitalized because of an abnormal lung shadow. The chest film provided evidence of pulmonary nodules. She was asymptomatic and had no labolatory abnormalities. Exploratory excision was performed to further assess the pulmonary nodules. The initial diagnosis was a suspected "hemangiopericytoma". She received combination chemotherapy but it was not effective. The nodules increased and decreased in size and number over the next decade from 1982, reaching a maximum by 1990 and a minimum by the end of 1992. However, her clinical condition remained stable during this period. Retrospective immunohistochemical analysis of CD10 and progesterone receptors confirmed a diagnosis of pulmonary parenchymal endometriosis. This was an unusual case of pulmonary parenchymal endometriosis that demonstrated various changes in the size and number of nodules without symptoms. There are few previous reports that demonstrated such nodular variation without symptoms over the course of disease.     

A case of true pulmonary carcinosarcoma]

The patient was a 47-year-old man who had visited a nearby physician with a chief complaint of bloody sputum. This patient underwent partial resection of the left upper lobe including a tumor adjacent to a huge bulla revealed by a plain chest radiograph. The resected tumor was composed of adenocarcinomatous and sarcomatous components, including partially-differentiated chondrosarcoma and rhabdomyosarcoma. The diagnosis was therefore true pulmonary carcinosarcoma. The sarcoma component recurred in the remaining part of the left lung. This recurrent lesion was surgically unresectable, but was regionally controlled with radiotherapy and bronchial arterial infusion of anticancer agents. However, distant metastases occurred to the brain, liver, and small intestine, and digestive tract bleeding also occurred. Nevertheless, chemotherapy, linac radiosurgery, percutaneous radiofrequency ablation, and partial small intestinal resection combined with rectal resection were performed. At autopsy, a metastatic lesion was also detected in the pancreas. Liver metastatic lesions were determined to be the adenocarcinomatous component, and the other recurrent or metastatic lesions, except for those in the brain, were all composed of poorly-differentiated sarcomatous tissue.     

A case of paracoccidioidomycosis with severe adrenal insufficiency

Paracoccidioidomycosis (PCM) is the most common systemic fungal disease in central-south America, but is rare in Japan. We experiensed a case of PCM in a patient, who came from Bolivia and presented with mouth pain and reduced dietary intake but no fever. Adrenal insufficiency was diagnosed with extremely high serum adrenocorticotropic hormone (ACTH) and was resolved with hormone supplementation. The PCM was treated with trimethoprim-sulfamethoxazole which was switched to itraconazole and improvement was achieved.     

Gastric paracoccidioidomycosis. A case report and considerations on the pathogenesis of this disease]

The authors report an unusual case of fungal gastric lesion in a patient with the chronic form of paracoccidioidomycosis. For a period of 8 months the major symptoms of the patient were abdominal pain and body weight loss. The endoscopic evaluation of the upper gastrointestinal tract showed a partial gastrectomy made previously, a great and irregular ulcer in the stomach and a granulated aspect of the duodenal mucosa. A granulomatous process and Paracoccidioides brasiliensis were observed in the histopathological examination of the gastric and duodenal biopsy tissue. New endoscopic evaluation 4 months after treatment with sulfadiazine revealed healing of the gastric ulcer. It was suggested that abdominal lymph nodes involvement, duodenal infection and anatomical and atrophic alterations of the stomach were predisposing factors for the gastric paracoccidioidomycosis.     

Micafungin therapy for a case of chronic necrotizing pulmonary aspergillosis]

The patient was a 42-year-old man who visited a physician with fever, and was diagnosed with pulmonary abscess. Antibiotic therapy was ineffective, and he was referred to our hospital. Chest CT scanning revealed a lesion with cavity formation with an infiltrative shadow in the right upper lobe, and another infiltrative shadow in the left upper lobe. Chronic necrotizing pulmonary aspergillosis (CNPA) was diagnosed on the basis of positive culture of bronchial lavage specimens and positive serological test results for Aspergillus, in addition to the clinical and radiographic features. Intravenous administration of micafungin (MCFG) was initiated with combination therapy of percutaneous cavity drainage, inhaled amphotericin B and oral itraconazole. Clinical symptoms and findings gradually improved, and he was discharged after 40 days of MCFG therapy. MCFG was safe and effective therapy in this case, and may be considered a new therapeutic option for CNPA.