[Birt-Hogg-Dubé syndrome with repeated pneumothorax; report of a case].

A 36-year-old man had been treated by thoracoscopic bullectomy because of left pneumothorax at another hospital. 2 weeks post-surgery, he was transferred to our hospital because of recurrent left pneumothorax. Chest computed tomography (CT) revealed multiple, irregular-shaped pulmonary cysts of various sizes predominating in the apex of both lungs. Thoracoscopy showed diffuse multiple thinwalled small cysts on the visceral pleura. Small cyst with air leakage was sutured and other cysts were cauterized. He was diagnosed as having Birt-Hogg-Dubé (BHD) syndrome by DNA sequence analysis of his FLCN gene.

[Adenocarcinoma of the lung in a young adult].

We report a case of 24-year-old young man with lung adenocarcinoma. The patient had been treated malignant lymphoma when he was 5 years old. The patient was pointed out a tumor shadow of the right lung at his 2008 health check, but he did not receive further examination. He was again pointed out a same tumor at his 2009 health check. Plain chest computed tomography (CT) revealed a nodule in S8 of right lung. A diagnosis of lung adenocarcinoma was established by the cytology specimens obtained with bronchoscopic examination. The patient underwent right lower lobectomy with systematic lymph node dissection. Pathological stage was pT2N2M0 stage IIIA. Lung cancer patients aged under 30 years old are rare. Lung cancer should be considered even in young patients with abnormal chest shadows.

'Lung age' predicts post-operative complications and survival in lung cancer patients.

BACKGROUND AND OBJECTIVE: The Japanese Respiratory Society recently proposed 'lung age' as an easily understood concept of respiratory function. In this study, we evaluated whether 'lung age' could be a useful predictor of post-operative respiratory complications and survival patients with lung cancer treated surgically. METHODS: The study recruited 308 patients who underwent surgery for primary non-small-cell lung cancer. All patients had preoperative pulmonary function testing. 'Lung age' was determined using the methods advocated by the Japanese Respiratory Society. Based on the difference between 'real age' (R) and 'lung age' (L), patients were classified into five groups: group A: R-L > 15 (n = 37), B: 5 < R-L R-L (n = 94). Clinicopathological factors, post-operative respiratory complications and survival were compared between the groups. RESULTS: Gender, smoking status and index, histology, operative approach and FEV(1) were significantly associated with the group classification. The incidence of complications was significantly higher in group E compared with other groups (P = 0.003). Multivariate analysis showed that the group classification by 'lung age' was an independent predictor of post-operative respiratory complications (P = 0.02). Overall survival differed significantly between the groups (P = 0.03). CONCLUSIONS: 'Lung age' could be useful for the prediction of post-operative respiratory complications and survival in patients with lung cancer treated surgically.

Spontaneous regression of lung adenocarcinoma: Report of a case.

This report presents a rare case of lung adenocarcinoma accompanied by partial spontaneous regression (SR). A 69-year-old woman with an abnormal shadow on chest X-ray was admitted to the hospital. She was diagnosed to have lung adenocarcinoma with right adrenal metastasis based on a thorough examination. However, a month after the diagnosis, the primary and metastatic lesions markedly shrank on the following computed tomography scan and (18)F-fluorodeoxyglucose positron emission tomograph. This was thought to be a partial SR of the cancer, and she was treated with a surgical procedure. She has since remained free of disease for 14 months after the surgery.

['Lung age' predicts post-operative complications and survival in lung cancer patients].

The Japanese Respiratory Society (JRS) recently proposed 'lung age' as an easily understood concept of respiratory function. In this study, we evaluated whether 'lung age' could be a useful predictor of post-operative respiratory complications and survival patients with lung cancer treated surgically. The study recruited 308 patients who underwent surgery for primary non-small cell lung cancer. All patients had pre-operative pulmonary function testing. 'Lung age' was determined using the methods advocated by the JRS. Based on the difference between real age' (R) and 'lung age' (L), patients were classified into five groups: group A: R-L > 15 (n = 37), B: 5 < R-L < or = 15 (n = 50), C: -5 < or = R-L < or = 5 (n = 73), D: -15 < or = R-L < -5 (n = 54), E: -15 > R-L (n = 94). Clinicopathological factors, post-operative respiratory complications and survival were compared between the groups. Gender, smoking status and index, histology, operative approach and FEV1 were significantly associated with the group classification. The incidence of complications was significantly higher in group E compared with other groups (p < 0.01). Multivariate analysis showed that the group classification by 'lung age' was an independent predictor of postoperative respiratory complications (p = 0.02). Overall survival differed significantly between the groups (p = 0.03). 'Lung age' could be useful for the prediction of post-operative respiratory complications and survival in patients with lung cancer treated surgically.

CD1a expression in PEComas.

According to the World Health Organization classification, neoplasms with perivascular epithelioid cell differentiation (PEComas) are mesenchymal tumors composed of histologically and immunohistochemically distinctive PEC. Generally, nearly all PEComas have immunoreactivity for both melanocytic (HMB-45 and/or melan A) and smooth muscle (actin (SMA) and/or desmin) markers. Recently the authors reported that benign clear cell sugar tumor of the lung, one of the PEComas, expressed CD1a. Therefore the purpose of the present study was to investigate the relationship between PEComas and CD1a expression. Nineteen PEComas were obtained, which included angiomyolipoma of the kidney or liver, lymphangiomyomatosis of the uterus or lung and clear cell sugar tumor of the lung. Eighteen tumors had alpha-SMA and HMB-45 expression and 16 had melan A expression. In contrast, all 19 tumors had CD1a expression. The present study confirms CD1a expression in many cases of PEComa. These data suggest that CD1a expression can be an additional new marker for PEComas and also supports the distinct and integrated disease entity of PEComas.

Pulmonary arteriovenous fistula with cerebral infarction successfully treated by video-assisted thoracic surgery.

We experienced a case in which a pulmonary arteriovenous fistula was found due to the occurrence of cerebral infarction. The patient was a 65-year-old female who had seen a local doctor for lightheadedness occurring upon rising in the morning. She underwent a cerebral MRI and was diagnosed to have a cerebral infarction. Upon closer examination, a chest radiograph revealed an abnormal shadow and a three-dimensional computed tomography (3D-CT) chest angiography detected a pulmonary arteriovenous fistula 32x30 mm in size with the feeder blood vessel A10 and drainer blood vessel V10 in the left inferior lobe S10, which was considered to be the cause of the cerebral infarction. Video-assisted thoracic surgery (VATS) was conducted for a segmental resection of the basal segment of the left lung. The patient's postoperative progress was good and there was no reoccurrence of the cerebral infarction. The rate of occurrence of cerebral infarction along with pulmonary arteriovenous fistula is considered to be 11.4%; however, cerebral infarction may be an early indicator of pulmonary arteriovenous fistula, and therefore, due attention must be paid.

Primary adenocarcinoma of the trachea resected using percutaneous cardiopulmonary support (PCPS).

We encountered one very rare case of primary adenocarcinoma of the trachea. The patient was a 72-year-old woman who was hospitalized immediately following significant dyspnea and mental confusion. A computed tomography scan of her chest revealed a 14x13x11 mm tumor in the trachea. After establishing artificial respiration, a tracheal tubular resection was immediately performed using percutaneous cardiopulmonary support (PCPS). A postoperative pathological examination led to the patient being diagnosed with primary adenocarcinoma of the trachea, and she was treated with adjuvant chemotherapy. Since then, for approximately 1 year and 6 months we have detected no relapse.

Thoracoscopic resection with intraoperative use of methylene blue to localize mediastinal parathyroid adenomas.

We report a case of thoracoscopic resection of mediastinal parathyroid adenomas using methylene blue to localize the tumors during the operation. After methylene blue 4 mg/kg was injected intravenously, we easily identified methylene blue-stained parathyroid glands and successfully resected them with sufficient surgical margins. The use of methylene blue for detection of parathyroid adenoma is a useful technique.

Sarcoid-like reaction in Cryptococcus neoformans infection.

We report a patient with Cryptococcus (C.) neoformans infection, who developed a case of sarcoid-like reaction (SLR). There have been reports of SLRs associated with malignancies. Although differentiating sarcoidosis from SLR is difficult, the patient was diagnosed as SLR because propionibacterium acnes bacterial (PAB) antibody staining of biopsy specimens was negative and the chest radiological findings improved after antifungal treatment. To our knowledge, this is the first report of SLR occurring during cryptococcal infection, and we believe that cryptococcal infection should be considered as a potential cause of SLR.

Prognostic implication of lymphatic vessel invasion in stage IB (pT2aN0M0) non-small cell lung cancer.

PURPOSE: To clarify the prognostic factors for recently revised stage IB non-small cell lung cancer. METHODS: Twenty-six specimens of completely resected stage IB (pT2aN0M0) non-small cell lung cancer were studied. Clinicopathological profiles of the 26 patients, including age, sex, smoking status, tumor size, histology, pleural invasion, and lymphatic and blood vessel invasion, were assessed. Correlation between tumor recurrence and these factors was assessed by statistical analysis. RESULTS: Lymphatic vessel invasion was the only statistically significant factor for postoperative tumor recurrence as assessed by multivariate analysis. Kaplan-Meier survival curves showed a 5-year recurrence-free rate of 77.8% in 26 patients with no lymphatic vessel invasion compared with 28.6% in those with lymphatic vessel invasion. CONCLUSION: Lymphatic vessel invasion of cancer cells was indicated as an independent prognostic factor in revised stage IB non-small cell lung cancer.

Thoracoscopic resection of a thoracic duct cyst that developed during follow-up for a thymic cyst.

A 29-year-old man had been diagnosed with an anterior mediastinal cyst 6 years earlier and was undergoing follow-up. At a follow-up visit, a newly developed cystic lesion was found in the middle mediastinum; therefore, the anterior mediastinal cyst and the middle mediastinal cyst were resected by thoracoscopic surgery. It was observed that the middle mediastinal cyst originated from the thoracic duct, and the thoracic duct was clipped. Pathologically, the diagnosis was a thymic cyst of the anterior mediastinum and a thoracic duct cyst of the middle mediastinum. The patient developed chylothorax after surgery, and a second thoracoscopic operation was performed. It revealed that part of the clipping of the caudal thoracic duct was incomplete, and leakage of chyle was observed. Hence, the clipping was performed again. The course after the second surgery was good. Thoracic duct cysts are rare even among mediastinal cysts and thus require caution due to their tendency to expand.

Sister Mary Joseph's nodule that originated from lung adenocarcinoma.

Umbilical metastasis of cancer, known as Sister Mary Joseph's nodule (SMJN), is a rare phenomenon. It is usually due to intraabdominal malignancies and is quite rare from lung cancer. Here we describe a case of SMJN that originated from advanced lung adenocarcinoma. SMJN should be noted as an important sign of some hidden malignancy including lung cancer.

Surgical outcome of video-assisted thoracic surgery for acute thoracic empyema using pulsed lavage irrigation.

PURPOSE: The essential points of video-assisted thoracic surgery (VATS) for acute thoracic empyema are the decortication of thickened pleura, resection of necrotic tissues and fibrin blocks, and drainage. Pulsed lavage irrigation, which is commonly used in orthopedic surgery as a method of sufficiently performing the technique, was used under a thoracoscope to study the efficacy of the treatment for acute thoracic empyema. METHODS: The subjects comprised 31 patients who had undergone VATS for acute thoracic empyema. There were 26 men and 5 women with an average age of 60.5 years. For the surgical technique, the thickened pus-producing pleura were decorticated under a thoracoscope. The pulsed lavage irrigation system was used after the intrathoracic space had become a single cavity. Using the tip for an intraspinal space, lavage and suctioning were repeated with 5-10 l of a pressurized warm saline solution. RESULTS: Fibrin blocks and necrotic tissues were easily removed by spray washing with pressurized fluid. The operating time was 150.8 min; the amount of bleeding, including suctioned pleural effusion, was 478.5 g; and the postoperative duration of drainage was 10.7 days. During the postoperative course, the addition of open window thoracotomy due to the relapse of empyema due to methicillin-resistant Staphylococcus aureus was observed in only one patient (3.2%). All of the other patients improved despite their concomitant diseases. CONCLUSION: The use of pulsed lavage irrigation under a thoracoscope for acute thoracic empyema provides simple, efficient débridement or drainage.

Pulmonary mucinous cystadenoma: a rare benign tumor of the lung.

Pulmonary mucinous cystadenoma (PMCA) is rare, with few reported cases. This tumor is histologically characterized by a benign proliferation of mucin-producing epithelial cells and bulky mucin inside the tumor. We present the case of a 71-year-old woman with increasing mass shadow on chest radiography who underwent tumor resection by video-assisted thoracic surgery. The tumor was diagnosed histologically as PMCA.

Thoracoscopic extended thymothymectomy for myasthenia gravis with aplastic anemia.

It is very rare for both myasthenia gravis and aplastic anemia to be complicated with a thymoma. A 74-year-old female was diagnosed to have aplastic anemia with pancytopenia and systemic myasthenia gravis with severe restrictive respiratory dysfunction. Chest CT showed a 5-cm diameter thymoma. After platelets and packed red blood cells were transfused before surgery, an extended thymothymectomy was performed with a bilateral thoracoscopic approach. The thymoma was noninvasive, stage I, and was classified as B1 according to the World Health Organization classification. After the operation, the patient was managed on artificial ventilation with no complications. The myasthenia gravis remitted with the concomitant administration of steroids and immunosuppresants, but the aplastic anemia was not ameliorated. A thoracoscopic thymothymectomy for such a high-risk case of infection and respiratory distress is appropriate surgical procedure, but the complications associated with aplastic anemia are intractable.

Subpleural intrapulmonary lymph node metastasis from colorectal cancer.

A 67-year-old male was admitted to our hospital because of pulmonary metastasis from colo-rectal cancer. Chest computed tomography revealed three tumor shadows just under the pleura. Because the tumors were adjacent to each other, we performed an en bloc pulmonary resection. Pathologically, two of the tumors were diagnosed as pulmonary metastases; the remaining tumor, however, was diagnosed as a subpleural intrapulmonary lymph node (IPLN) metastasis. Metastasis to the IPLN is rare, but when the IPLN is located adjacent to a pulmonary metastasis, as observed in this case, it may be possible.

The use of Blake drains following general thoracic surgery: is it an acceptable option?

As a method of chest drainage, we analyzed the extended utility of silastic flexible drains (Blake drains, Ethicon, Inc., Somerville, NJ) for general thoracic surgery. In 420 cases of general thoracic surgery, Blake drains were used. To examine the utility of Blake drains, we investigated the diseases for which they were used, their effectiveness in addressing postoperative complications. The treated diseases for which Blake drains were used comprised 181 cases of primary lung cancer, 44 cases of metastatic lung tumor, 57 cases of benign lung disease, 32 cases of mediastinal tumor, 6 cases of myasthenia gravis, 76 cases of spontaneous pneumothorax, 14 cases of chest wall and/or pleural tumor, 6 cases of empyema, and 4 cases of diaphragmatic disease. Blake drains functioned efficiently in 3 cases of re-operation for postoperative bleeding, 2 cases of adhesion therapy with drugs for persistent air leaks, and 1 case of re-operation for chylothorax. There were no cases of either complications or patient complaints of discomfort resulting from drain placement. The use of Blake drains for general thoracic surgery is considered to be an acceptable option, and it is necessary to proceed with further investigations of larger numbers of cases.

Mediastinal lymph node carcinoma of an unknown primary site: clinicopathological examination.

PURPOSE: We examined the clinicopathological features of four mediastinal lymph node carcinomas from an unknown primary site. METHODS: Four patients with mediastinal lymph node carcinoma from an unknown primary site were treated at our hospital during the past 6 years. RESULTS: All of the patients were men, with an average age of 74.0 years (range 70-78 years). Histologically, one lesion was poorly differentiated adenocarcinoma, two were poorly differentiated squamous cell carcinoma, and one was undifferentiated carcinoma. Radical resection of the mediastinal tumor and radiotherapy were performed in two patients, both of whom were still alive with no evidence of recurrence at 44 and 82 months, respectively, after the operation. Thoracoscopic biopsy was performed in two patients, and both underwent chemoradiotherapy. These two patients were alive at 24 and 33 months, respectively, after the biopsies. CONCLUSION: Radical resection of mediastinal lymph node carcinoma with an unknown primary site has the possibility of a good prognosis. In nonradical cases, it is important to perform a biopsy to make an accurate diagnosis and then administer radiotherapy and/or chemotherapy postoperatively.

A small alveolar adenoma resected by thoracoscopic surgery.

Alveolar adenoma (AA) is a rare lung benign tumor originated from type II pneumocytes. It presents as a well-defined nodule in clinical images, but is difficult to differentiate from early-stage lung cancer. We treated a 58-year-old woman with a small-sized AA measuring 8 x 6 mm in the upper lobe of the left lung by performing a thoracoscopic resection. The AA does not cause any symptoms and it is often incidentally detected as an abnormal shadow. There have been no reported cases of recurrence after a resection of AA, but a subsequent increase in such cases is expected, and we believe that it is necessary to understand the characteristics of this typical benign lung tumor.