[Subacute cor pulmonale due to microscopic pulmonary tumor embolism].

We report a case of subacute pulmonary hypertension caused by microscopic pulmonary tumor embolism due to the dissemination of gastric cancer cells. The patient, a 61-year-old man with no history of malignant diseases, was admitted to our hospital on October 14 in 1998 because of cough and dyspnea on effort, that had developed since the previous month. On admission, chest radiography including CT scans showed slight cardiomegaly and disseminated reticulonodular shadows predominating in the lower lung fields of both lungs, and arterial blood gas analysis disclosed severe hypoxemia. Lung perfusion scintigraphy revealed multiple irregular defects in both lungs. Echocardiography indicated right ventricular overload, and the pulmonary artery systolic pressure was estimated to be higher than 80 mmHg. Disseminated intravascular coagulation (DIC) developed on the 6th day of hospitalization. Multiple pulmonary embolism with DIC of unknown cause was diagnosed, and the patient was given anticoagulant therapy with heparin. However, he died of respiratory failure on the 7th day of hospitalization. At autopsy, an invasive cancer was found in the stomach, resembling type IIc early gastric cancer. The lumens of the pulmonary arterioles were significantly narrowed by fibrocellular proliferation and thrombi accompanying tumor cell clusters, and some of the microvessels were completely occluded. Disseminated microscopic pulmonary metastasis of malignant tumors should be included in the differential diagnosis of subacute pulmonary hypertension due to multiple pulmonary embolism of unknown cause.

[Pulmonary Nocardia otitidis-caviarum infection in a patient with Cushing's disease].

While Nocardial infections are being diagnosed with increasing frequency, infection with Nocardia otitidiscaviarum remains relatively uncommon. We report a case of pulmonary Nocardia otitidis-caviarum infection in a 35-year-old man with Cushing's disease. This work describes the first case of nocardiosis in Japan caused by Nocardia otitidis-caviarum in Cushing's disease. The patient was admitted to our department because of edema. A diagnosis of Cushing's disease was made on the basis of elevated serum levels of cortisol and adrenocorticotropic hormone (ACTH) and pituitary adenoma was found in a cranial CT scan. One month after admission, chest radiographs showed a large bilateral mass on the lung fields. Nocardia otitidis-caviarum was isolated from the sputum. The patient responded poorly to intravenous PAPM/BP, but later improved after treatment with trimethoprim-sulfamethoxazole, but he died of heart failure and respiratory failure after the initiation of this therapy. This case demonstrated that nocardiosis must be considered in differential diagnosis as an opportunistic infection.

[Sarcoidosis in a man and his identical twin sons].

Sarcoidosis is known to be a disease characterized by high familial aggregations. We reported on the genealogy of sarcoidosis in a man and his identical twin sons. The patient we encountered was a 49-year-old man admitted to our hospital because of abnormal chest X-ray findings observed during a medical checkup. Cervical and hilar lymph nodes were swollen on both sides, and lesions were observed in the right lung field. Biopsy specimens from cervical lymph nodes disclosed epithelioid cell granuloma without caseous necrosis. A diagnosis of sarcoidosis was made. The disease had also been diagnosed in the patient's identical twin sons 2 years earlier. They had been successfully treated and no recurrence has been observed. HLA typing disclosed that the father and his twin sons had identical A 33 (19), B 44 (12), DR 2, and DR 12 genes. This finding suggested that genetic factors are involved in the onset of this disease.

[Mixed connective tissue disease-associated interstitial pneumonia complicated by pneumomediastinum during prednisolone therapy].

A 59-year-old woman was admitted to our hospital with exertional dyspnea. Linear and reticular opacities in the middle and lower fields of both lungs were observed on chest roentgenograms and chest computed tomograms (CT). The presence of anti RNP-antibody and Raynaud's phenomenon, sclerosis of the fingers, and leukopenia yielded a diagnosis of mixed connective tissue disease associated with interstitial pneumonia. The symptoms and lung involvement were alleviated after the administration of prednisolone (40 mg/day). However, the patient experienced the sudden onset of dyspnea during pulmonary function tests. A chest X-ray film disclosed mediastinal air around the left pulmonary artery, and a chest CT scan demonstrated some blebs in the left lower lung field. After tapering the dosage of prednisolone in 5 mg increments per week, the pneumomediastinum disappeared without treatment. The clinical features and laboratory data findings suggested the patient's interstitial pneumonia was associated with systemic lupus erythematosus rather than with progressive systemic sclerosis or dermatomyositis. The pneumomediastinum may have been due to the rupture of blebs secondary to interstitial pneumonia during pulmonary function tests or as a result of steroid therapy.

Type 1 diabetes mellitus caused by treatment with interferon-beta.

A 57-year-old man was referred to our outpatient clinic after interferon-beta (IFN-beta) treatment for 7 weeks. While IFN-beta therapy was continued in our outpatient clinic, his blood glucose level increased gradually, and he was admitted to our hospital for hyperglycemia. The patient was prescribed a 1,600-kcal diet and intensive insulin therapy was performed. GAD antibody became positive 15 months after the start of IFN therapy, and disappeared 27 months after the start of IFN therapy. Insulin secretion was depleted and the patient had HLA-DR4, B54, and DRB1*0405. This appears to be a case of type 1 diabetes mellitus induced by administration of IFN-beta alone.

[Three cases of semi-invasive pulmonary aspergillosis occurring in healthy or diabetic individuals].

We encountered three cases of suspected semi-invasive pulmonary aspergillosis, occurring in healthy or diabetic patients. Radiographic findings disclosed cavity formation in 2 of the patients, but only nodular lesions in 1. The beta-D glucan level was elevated in 1 patient, but was within the normal range in the other 2. Morphological findings indicated that all 3 patients had pulmonary aspergillosis. Following the oral administration of FLCZ to 1 patient and ITCZ to the other 2 for about 2 months, the radiographic lesions resolved and hematologic inflammatory reactions cleared up. Gefter et al and Sider et al have defined 2 types of aspergillosis that occur in mildly immuno-compromised or healthy individuals: the semi-invasive type, which destroys pulmonary tissue without vascular invasion and leads to cavity formation; and the locally invasive type, which is characterized by nodular or massive radiographic lesions without cavity formation. Semi-invasive aspergillosis was diagnosed in all 3 of the cases we reported.

Cancer-associated retinopathy during treatment for small-cell lung carcinoma.

A 70-year-old woman with small-cell lung carcinoma (c-T4N2M0) was treated by six courses of combination chemotherapy (carboplatin and etoposide). After two weeks, she complained of a sense of darkness and night blindness. A Western blot analysis showed that the patient's serum bound with the recombinant 23-kDa retinal cancer-associated retinopathy (CAR) antigen at 1:1,000 dilution. Her visual acuity became so poor that she could only recognise a hand motion at 50 cm despite treatment with corticosteroids and combination chemotherapy. The patient was diagnosed as having a rare type of CAR because CAR is usually found before the diagnosis of primary cancer.

[Sarcoidosis synchronously detected in identical twins].

We report on the synchronous detection of sarcoidosis in identical 22-year-old male twins. The patients visited a nearby physician because of fever and cough, were found to have abnormal chest shadows and thus admitted to our hospital. Both had swollen cervical and hilar nodes and lesions in both left and right lung fields, and also presented similar clinical symptoms. However, bilateral uveitis was observed only in the elder twin. Because epithelioid cell granulomas without caseous necrosis were histopathologically observed in tissue specimens from their cervical lymph nodes, the patients were given a diagnosis of sarcoidosis. Although they were positive for the HLA-D antigens DR 2 and DR 12, they were negative for DRw 52. an antigen that is considered to be quite common in Japanese sarcoidosis patients. Genetic factors were thought to be involved in the onset of sarcoidosis in these twins.

[Chronic thromboembolic pulmonary hypertension associated with initial pulmonary involvement in Takayasu arteritis].

A 37-year-old non-smoking woman was admitted to our hospital because of nocturnal coughing and exertional dyspnea after using an ultrasonic humidifier. Chest roentgenograms and computed tomography showed many ground-glass opacities in the right upper lobe and left upper lung field. Her symptoms were alleviated and erythrocyte sedimentation rate (ESR) and C-reactive protein (CRP) levels returned to normal following the administration of prednisolone (40 mg/day). The same symptoms subsequently recurred, and ESR and CRP levels increased proportionately to a decreased dosage of prednisolone. In addition, chest computed tomography showed new opacities in the left lower lobe. Transbronchial lung biopsy specimens revealed multiple stenotic or obstructive changes in the small pulmonary arteries by organizing thromboemboli, with no vasculitis. The diagnosis of chronic thromboembolic pulmonary hypertension was made on the basis of multiple defects from lung perfusion scintigraphy, irregular or tapering of the pulmonary arteries detected by pulmonary arteriograms, and increased pulmonary pressure measured by echocardiography. We propose that the pulmonary thromboembolism observed in this case was related to the initial pulmonary involvement of Takayasu arteritis, since the patient's symptoms, ESR and CRP levels all improved markedly following the administration of prednisolone.