Treatment of pulmonary sporotrichosis

Pulmonary infection with Sporothrix schenckii is extremely rare. The small number of reported cases and lack of comparative clinical trials make assessment of treatment regimens difficult. Three modalities should be considered: Saturated solution of potassium iodide (SSKI), amphotericin B, and resective surgery. Available data suggest that SSKI is unlikely to be successful in cavitary disease or disease in immunocompromised hosts. Amphotericin B alone has been successfully used to cure cavitary pulmonary sporotrichosis, but overall cure rates probably are no higher than 50%, and the probability of cure in bilateral apical disease is dismal. Surgical resection has been successful when used alone in a small number of cases. The highest cure rates (70% to 80%) appear to be a result of management with amphotericin B or SSKI combined with resective pulmonary surgery. The dosage and duration of amphotericin B or SSKI to be used in conjunction with surgical resection is unknown.     

Primary pulmonary sporotrichosis.

A 34-year-old alcoholic and drug addict developed cavitary pulmonary sporotrichosis that progressed slowly during 6 years. Pulmonary resection and pre- and postoperative therapy with amphotericin B were associated with prompt clinical improvement with no evidence of relapse during a 2-year follow-up. Histologic examination of lung revealed granulomatous inflammation with organisms consistent with Sporothrix schenckii, and interstitial talc (magnesium silicate) granulomas. The latter finding was consistent with the history of intravenous drug abuse. Although the presence of silicates in lung enhances the pathogenicity of some microorganisms, the relation of these findings to the pathogenesis of sporotrichosis in our patient is unclear.     

Treatment of pulmonary sporotrichosis with ketoconazole

Antifungal therapy that included ketoconazole failed in a 44-year-old woman with pulmonary sporotrichosis progressing slowly over a seven-year period. On the basis of this case and the modest amount of experience reported in the literature, ketoconazole does not appear to be effective in the treatment of pulmonary sporotrichosis.     

A case of primary pulmonary cryptococcosis showing spontaneous recovery

A 37-year-old woman was admitted with fever and cough. The chest roentgenogram revealed a solitary large round mass in the right lower lung field. Diagnosis was made by transbronchial lung biopsy. There was no evidence of abnormal cell-mediated immunity. The patient made an uneventful recovery and the chest X-ray showed progressive clearing without specific therapy. After 6 months, the mass lesion disappeared. This case suggests that specific therapy may not be necessary for all cases of primary pulmonary cryptococcosis.     

A case of sarcoidosis with primary pulmonary cavitation]

A 30-year-old woman presented with multiple nodular shadows which enclosed a cavity on a chest radiograph. Chest computed tomographic (CT) images showed mediastinal lymphadenopathy, and multiple nodular opacities enclosing a cavity. Histopathological findings of biopsy specimens from the lung and mediastinal lymph nodes revealed noncaseating epithelioid cell granulomas without any evidence of Mycobacterium or fungal growth. The lesion in the lung included granulomatous vasculitis. Even without corticosteroid or any other therapy, the lung lesions resolved and the cavity disappeared. We report a case of sarcoidosis with primary acute cavitation.     

A case of preoperatively diagnosed primary pulmonary leiomyosarcoma]

A 29-year-old woman had been suffering from right back pain for 3 months. Chronic pulmonary thromboembolism was suspected and she was referred to our hospital. She presented with no risk factors for thromboembolism, and during the previous 6 months had lost 4 kg in body weight. Chest radiography showed nodular shadows in the lower field of the right lung. Contrast-enhanced computed tomography demonstrated a filling defect in the right pulmonary artery and nodular lesions in the lower field of the right lung, which were considered to be signs of pulmonary infarction. Absence of perfusion into the right lung was demonstrated by a perfusion scan. Right heart catheterization showed normal pressure in the pulmonary arteries, and pulmonary angiography showed an abrupt cutoff of the right pulmonary artery, which was similar to the finding of pulmonary thromboembolism. A transvenous catheter suction biopsy was performed in the right pulmonary artery and the histopathologic findings yielded a diagnosis of leiomyosarcoma. The patient underwent surgical resection under total cardiopulmonary bypass. A large tumor completely filled the right main pulmonary artery and invaded the posterior wall of the pulmonary trunk close to the left main pulmonary artery. Primary pulmonary leiomyosarcoma is a rare tumor and its prognosis is very poor. Radical surgical resection is the only effective treatment, but early diagnosis is very difficult. Transvenous catheter suction biopsy is a useful procedure for the early diagnosis of pulmonary artery sarcoma.     

A case of primary pulmonary artery myxosarcoma associated with severe pulmonary hypertension]

A 50-year-old man presented with progressive dyspnea on exertion, but with no history of chest pain or syncope. Chronic pulmonary thromboembolism was suspected and he was referred to our hospital. On ausculation, a grade 3 systolic murmur was heard, that was loudest in the fifth intercostal space lateral to the right sternal border. Chest radiography showed mild cardiomegaly and ventilation-perfusion scan revealed absence of perfusion in the left lung and the upper field of the right lung. Contrast-enhanced helical CT showed large mural defects in both main pulmonary arteries, clearly delineated by contrast medium. The left pulmonary artery was nearly completely occluded, and eccentric defects were observed projecting into the lumen of the pulmonary trunk. A tumor originating in the pulmonary artery was suspected, but a definitive diagnosis of the mass could not be made with pulmonary angiography and magnetic resonance imaging. The mean pulmonary arterial pressure was 50 mmHg. Further radiologic examinations failed to reveal the source of the embolus or tumor. It was decided to attempt surgical excision under total cardiopulmonary bypass. At operation, a gelatinous, lustrous, yellowish mass was found partially occluding the right main pulmonary artery and completely occluding the left. The tumor adhered tightly to the intima of the vessel and was inoperable. The patient could not be weaned from percutaneous cardiopulmonary support and died 3 days after surgery. Histologic examination of the excised specimen revealed myxosarcoma.     

A case of primary leiomyosarcoma of the pulmonary artery]

A 74-year-old woman was admitted to our hospital because of right chest pain. The chest radiograph showed right hilar pulmonary artery dilatation. A mass exhibiting low intensity in T1-weighted images and high intensity in T2-weighted images was disclosed in the right pulmonary artery. Because of its clinical course and the MRI findings, the mass was thought to be tumorous tissue, and so pneumonectomy was performed. Leiomyosarcoma was diagnosed from the histological findings. Primary artery sarcoma is rare and the prognosis is considered to be extremely poor. The patient was successfully treated and had a good clinical outcome.     

A case of primary fibrosarcoma of the pulmonary artery]

A 38-year-old-man was admitted to our hospital complaining of exertional dyspnea. Chest radiography and CT showed a huge mass that completely occluded the left main pulmonary artery and spread into the right main pulmonary artery. The tumor was diagnosed as sarcoma by transbronchial biopsy. Radiation therapy obtained temporary partial response. He died of respiratory insufficiency after 14 months. The autopsy revealed primary pulmonary artery sarcoma: fibrosarcoma arising from the left pulmonary artery. To our knowledge, this is the first case report of fibrosarcoma of the pulmonary artery in Japan.     

Multifocal systemic sporotrichosis with lobar pulmonary involvement

Multifocal systemic sporotrichosis (disseminated sporotrichosis) with lobar pulmonary involvement is uncommon. We describe successful treatment with amphotericin B in such a patient and review data from 1 other similar case previously reported and 7 with nonlobar pulmonary involvement.     

原发性肺动脉肉瘤1例并文献复习

目的 提高对原发性肺动脉肉瘤的认识和诊断水平。方法 结合我院1例经手术病理证实的原发性肺动脉肉瘤患者的临床资料与国内外有关文献,对原发性肺动脉肉瘤的临床表现、影像学表现、实验室检查结果进行分析。结果 原发性肺动脉肉瘤的临床表现与肺动脉血栓栓塞相似,应用CT肺动脉造影鉴别有一定局限性,正电子发射型计算机断层扫描与CT合用有助于诊断。结论 原发性肺动脉肉瘤临床表现及常规影像特异性较少,正电子发射型计算机断层扫描与CT结合有更大的诊断价值。     

A case of sarcoidosis with primary acute pulmonary cavitation]

A 23-year-old man was admitted to our hospital on June 24, 1991, because of worsening chest X-ray findings of sarcoidosis. In August 1990, he was referred to our outpatient office, because of BHL and nodular lesions on chest X-ray film performed at his company 4 months earlier. At that time, serum ACE was elevated to 34.0 IU/l, and Ga scintigraphy showed abnormal uptake in bilateral lacrimal and salivary glands, mediastinal and hilar lymph nodes, and in the lung fields. TBLB specimen showed noncaseating epithelioid granuloma with giant cells and negative stains for acid-fast bacilli. Although it was planned to follow this patient without medication, he did not return to our outpatient department. In June 1991, because of worsening of lesions in the lung at annual checkup at his company, he was referred and admitted for steroid therapy. Chest X-ray film on admission showed BHL, multiple nodular lesions in both lung fields, and bullous change in the left upper lobe. Chest CT on admission showed three cavitating lesions within preexisting nodules. PPD skin test was negative, and sputum smears and cultures were repeatedly negative for pyogenic bacteria and acid-fast bacilli. Therapy was initiated with prednisolone 30 mg daily. Four months later, there was marked resolution of BHL and nodular lesions, and the cavitating lesions were no longer visible on chest X-ray film. From the clinical and radiological observations, it is concluded that the cavitating lesions in the present case were primary acute pulmonary cavitation in sarcoidosis, distinct from infection, bullae, or cystic bronchiectasis which are seen in the chronic and fibrotic stages of sarcoidosis.     

A case of primary pulmonary proteinosis with bilateral pneumothorax]

A 40-year-old man was admitted to our hospital because of chronic cough and mild exertional dyspnea. Radiographs of the chest showed diffuse infiltrative shadows in both lung fields. A diagnosis of pulmonary alveolar proteinosis (PAP) was made by examination with a flexible bronchoscope and bronchoalveolar lavage, with transbronchial biopsy. After diagnosis, the patient underwent whole-lung lavage with temporary improvement. Due to the recurrence of his illness, he needed a total of four whole-lung lavages over the course of his illness. However, the exertional dyspnea became progressively worse. Bilateral pneumothorax developed suddenly and led to his death. This case indicates the possibility that deterioration of PAP despite whole-lung lavage may sometimes be followed by pneumothorax.     

A case of primary macroglobulinemia with acute pulmonary edema]

A 54-year-old man with acute respiratory failure underwent emergency admission to our hospital. Acute pulmonary edema combined with severe pneumonia was strongly suspected, and treatment with diuretics, antibiotics, and corticosteroids was effective. The diagnosis, primary macroglobulinemia, was made from the following findings: a high level of the lgM 1 type monoclonal protein, and an elevated serum viscosity with characteristic "linked sausage effects" seen in the retinal veins. Lymphoplasmacytoid cells, plasma cells, and small lymphocytes were seen in the bone marrow and lymphoplasmacytoid cells were also seen in the smear of the patient's peripheral blood. The types of malignant cells found in his bone marrow were also seen in both infiltrative lesions in the lung parenchyma and the pleural effusion. We concluded that this was a case of primary macroglobulinemia with acute pulmonary edema, which was caused partly by the hyperviscosity syndrome. The antitumor effect of the corticosteroid might have some effect on the recovery from acute respiratory failure and primary macroglobulinemia. He was transferred to the Department of Hematology to receive suitable chemotherapy.