[Ectopic Adrenocorticotropic Hormone-Producing Pulmonary Carcinoid Presenting as Cushing's Syndrome after Intrapleural Hyperthermic Chemotherapy].

The patient was a woman in her 70's was referred to our hospital because of an abnormal shadow on chest roentgenogram at an annual medical checkup. Since preoperative examinations suggested lung cancer in the right middle lobe, thoracoscopic right middle lobectomy was planned. However, pleural dissemination was detected at surgery and we changed the treatment plan to the intrapleural hyperthermic chemotherapy. During the postoperative course, facial edema, hypokalemia, and hyperglycemia developed, and the diagnosis of Cushing's syndrome was suggested based on an increase in serum level of adrenocorticotropic hormone (ACTH) and cortisol, and was confirmed by a dexamethasone suppression test. Intrapleural hyperthermic chemotherapy was likely to collapse the ACTH-producing tumor leading Cushing's syndrome.

CT radiomics analysis of lung cancers: Differentiation of squamous cell carcinoma from adenocarcinoma, a correlative study with FDG uptake.

BACKGROUND AND PURPOSE: Pulmonary squamous cell carcinoma (SCC) was reported to have significantly higher histogram_kurtosis obtained by unenhanced computed tomography (CT) and a greater maximum standardized uptake value (SUVmax) determined by 18F-fluorodeoxyglucose positron emission tomography/computed tomography (18F-FDG-PET/CT) than pulmonary adenocarcinoma (AD). The first study aim was to investigate whether CT radiomics features could differentiate SCC from AD. The second aim was to investigate correlations between CT radiomics features and SUVmax. MATERIALS AND METHODS: Forty solid lung tumors < 3 cm in diameter pathologically proven to be SCC (n = 18) or AD (n = 22) were included. The SUVmax was determined by 18F-FDG-PET/CT and 42 CT radiomics features were obtained from unenhanced CT. A Mann-Whitney U test was used to compare the SUVmax and each CT radiomics feature to differentiate SCC from AD. The area under the curve (AUC) was calculated from receiver operating characteristic (ROC) analysis. A binary logistic regression analysis was performed for a combination of SUVmax with each CT radiomics feature. A Spearman rank correlation analysis was performed to determine correlations between SUVmax and CT radiomics features. RESULTS: Twenty-six of 42 CT radiomics features were significantly different between groups. The best AUC of the CT radiomics features was 0.81 for both the GLRLM_SRHGE and GLZLM_HGZE. The AUC value improved when the shape_sphericity feature was combined with SUVmax (AUC = 0.92). The CT radiomics features exhibiting strongest correlations with the SUVmax were different in both AD and SCC groups. CONCLUSION: CT radiomics features correlated with tumor structural characteristics of SCC and AD, and were closely related to metabolic information from FDG-PET/CT.

Radiation-induced sarcoma in a 10-year survivor with stage IV EGFR-mutated lung adenocarcinoma.

A 70-year-old Japanese man with stage IV EGFR-mutated lung adenocarcinoma complained of right mild back pain. The patient had been heavily treated with several cytotoxic or molecular targeted agents for 10 years and received a palliative radiation therapy of 2nd sacral vertebra 5 years ago. Computed tomography showed the abnormal lesion in right iliopsoas muscle. A pathological examination confirmed undifferentiated pleomorphic sarcoma, consistent with the diagnosis of radiation-induced sarcoma (RIS). Since RIS is a rare late-onset complication of radiation therapy, to our knowledge, this is the first report of RIS that was associated with advanced lung cancer and detected after palliative radiation therapy. The careful long-term follow-up is thus necessary even after palliative radiation therapy and we have to be aware of the existence of RIS.

Thirteen cases of pulmonary dirofilariasis in a single institution in Okinawa Island.

Pulmonary dirofilariasis is an infection caused by Dirofilaria immitis, which is an endemic parasite in Japan. We experienced 13 surgical cases of pulmonary dirofilariasis in our hospital. Of the 13 patients, 61.5% were men. The responsible lesions were located in the right lung in all cases, and 76.9% of them were in the lower lobe. Histologically, 12 cases showed necrotic nodules with peripheral granuloma with worms inside the pulmonary artery. One case did not show a necrotic nodule but showed only thickening and hyalinization of the pulmonary artery wall with a degenerated worm inside. Eosinophils were found histologically in all cases. Thirteen cases of dirofilariasis in one institution seem to be the largest number in Japan, based on previous reports. One reason for this increased prevalence may be the hot and humid climate of our prefecture considering the ecology of the mosquito as a vector. Elastic staining and eosinophils in peripheral granulomatous areas can contribute to the diagnosis when the worms are degenerated.

Comparison of GLUT-1, SGLT-1, and SGLT-2 expression in false-negative and true-positive lymph nodes during the 18F-FDG PET/CT mediastinal nodal staging of non-small cell lung cancer.

INTRODUCTION: Although positron emission tomography (PET) with 2-deoxy-2-[fluorine-18]fluoro-d-glucose integrated with computed tomography (CT), (18F-FDG PET/CT), has recently improved the mediastinal nodal staging of non-small cell lung cancer (NSCLC), this method can show false negativity. We immunohistochemically investigated the expression of glucose transporters (GLUT-1, SGLT-1, and SGLT-2) in false negative and true positive mediastinal nodes via 18F-FDG PET/CT. METHODS: We investigated patients with clinically-diagnosed N0/pathological N2 diseases and patients with clinically-diagnosed N2/pathological N2 disease. The patients who were included in this study were evaluated using 18F-FDG PET/CT followed by surgical resection between January 2004 and December 2015. The expression of GLUT-1, SGLT-1, and SGLT-2 in the metastatic mediastinal lymph nodes, and clinicopathological variables such as primary tumor size, lymph node size, histological type, and SUVmax of the primary lesion, were compared between false negative nodes and true positive nodes. RESULTS: The total number of PET false negative metastatic mediastinal lymph nodes was 22 in the 17 patients who were clinical N0/pathological N2, and the number of PET true positives was 15 in the 11 patients who were clinical N2/pathological N2. GLUT-1 expression was positive in five false negative nodes and 10 true positive nodes. SGLT-2 expression was positive in 12 false negative nodes and one true positive node, whereas both false negative and true positive nodes showed no SGLT-1 staining. Univariate analysis showed that the reduced expression of GLUT-1 (P = 0.015), and overexpression of SGLT-2 (P = 0.004) were the significant causative factors for false negative nodes. Multivariate analysis also showed that the reduced expression of GLUT-1 (P = 0.012) and overexpression of SGLT-2 (P = 0.006) were the significant causative factors for false negative nodes. CONCLUSION: It suggests that the reduced expression of GLUT-1 and overexpression of SGLT-2 are associated with false-negative lymph node metastases in NSCLC.

The Presence of Coexisting Lung Cancer and Non-Tuberculous Mycobacterium in a Solitary Mass.

BACKGROUND Whereas non-tuberculous mycobacterium (NTM) pulmonary disease can mimic lung cancer as a solitary pulmonary nodule or mass, the coexistence of lung cancer and NTM pulmonary disease in a single nodule or mass is rare. We report such a rare case, highlighting that during a bronchoscopes examination which comprises taking a transbronchial lung biopsy (TBLB), bronchial brushing, and bronchial lavage, a positive mycobacterium culture result for sputum or bronchial lavage fluid does not exclude the possibility of a concomitant lung cancer. CASE REPORT An 87-year-old male was referred to our institution for evaluation of an abnormal shadow on a chest x-ray scan. He had been previously healthy with no symptoms and an unremarkable medical history. A contrast-enhanced CT scan showed a cavitating mass measuring 20×40 mm with a thick ring-enhancing irregular wall in the left lower lobe. Although the TBLB of the lesion showed no malignant cells, sputum acid-fast bacilli smear and culture of the bronchial lavage fluid yielded positive results. An NTM infection, instead of lung cancer was suspected to have caused the mass because a Mycobacterium tuberculosis polymerase chain reaction showed negative results. However, we performed the surgery because NTM pulmonary disease and lung cancer cannot be differentiated. The results of a pathological examination of the mass showed an adenocarcinoma, and M. avium complex was detected in the cancer tissue culture. CONCLUSIONS Physicians should suspect the co-existent lung cancer and NTM infection in patients with solitary lung masses that yield a positive mycobacterium culture result for sputum or bronchial lavage fluid.

Mucoepidermoid Carcinoma of Arising from a Bronchogenic Cyst of the Diaphragm.

INTRODUCTION: Bronchogenic cysts may rupture or become infected, and malignant degeneration may occur. Although various types of malignant degeneration have been described, only a few reports of mucoepidermoid carcinoma arising from a bronchogenic cyst have been published. We report such a case. CASE: A 77-year-old female was referred to our institution for evaluation of left chest pain. A computed tomography scan showed an enhancing 65 × 70 mm mass of the left diaphragm. Based on the intraoperative findings of an intradiaphragmatic tumor involving the lower lobe of the left lung, the resection of the tumor with the wedge resection of left lower lobe and partial resection of the left diaphragm was performed. Histopathologic examination revealed a mucoepidermoid carcinoma arising from a bronchogenic cyst of the diaphragm with the presence of fibrous adhesion to the lower lobe. CONCLUSION: We believe that complete resection of any bronchogenic cyst is justified.

Postoperative Lung Torsion With Retained Viability: The Presentation and Surgical Indications.

BACKGROUND: We review our experience with postoperative lung torsion with retained viability. METHODS: A total of 2165 patients underwent pulmonary resection (lobectomy or segmentectomy) at our institution between 1 January, 1986, and 31 March, 2017. Eight (0.3%, six males and two females: median age, 68 years) had lung torsion with retained viability. RESULTS: The right upper lobe was resected in seven patients, while the left upper segment was resected in one patient. The lung torsion with retained viability was the right middle lobe in seven patients and the left lingular segment in one patient. A bronchoscopic examination was performed in four patients to diagnose the pulmonary torsion; however, it demonstrated no specific findings. Subsequently, computed tomography (CT) was performed in all the patients, and lung torsion was diagnosed in all the patients based on the CT findings. None of the patients showed any symptoms when lung torsion was diagnosed in them. The diagnosis of pulmonary torsion was made at a median of 4 days (range, 1-22 days) after the initial surgery. Six patients underwent detorsion of the affected lung, while one patient had a lobectomy, and one patient received conservative management. The lungs of all patients in which detorsion was performed adequately re-expanded. Frequent pneumonia in the viable torsed lung was diagnosed as a cause of death in the one patient who received conservative management. CONCLUSION: The timely decision to follow a surgical approach for lung torsion with retained viability can lead to a satisfactory outcome.

A rare case of congenital bronchoesophageal fistula in an adult.

INTRODUCTION: When congenital bronchoesophageal fistulas exist without atresia of the esophagus, the diagnosis can be delayed, although symptoms may occur early following fistula development. Therefore, while they are usually found in infants, they can be extremely rarely found in adults. We herein report a rare case of bronchoesophageal fistula without atresia in an adult. CASE: An 69-year-old male presented to the outpatient clinic with a decades-long history of cough with expectoration immediately after taking food, especially liquids. Computed tomograph, esophagoscopy, and esophagography revealed the fistulous communication between the mid-esophagus and right lower lobe bronchus, with consolidation in the right lower lobe. We performed right lower lobectomy with the closure and excision of the fistula. The histopathology of the fistula revealed the mucosa to be lined by stratified squamous epithelium. There was no evidence of inflammation, granuloma, or carcinoma. CONCLUSION: In conclusion, despite the benign nature of this malformation, if left untreated, it can cause long-term debilitating respiratory symptoms associated with the fistula. Therefore, the diagnosis should be considered in the evaluation of recurrent lung infection.

Giant pedunclated lipoma of the esophagus: A case report.

INTRODUCTION: Although Esophageal lipoma is extremely rare and pathologically benign, surgical excision of the lipoma is recommended when symptomatic or uncertain biological behavior. In general, some of the esophageal lipoma has a stalk. The pedunclated non-invasive tumor can be removed by stalk ligation, which is either endoscopic or surgical approache. Therefore, the preoperative evaluation is essential. We herein present a case of a huge esophageal lipoma. CASE REPORT: A 82-year-old man, with a wet cough and dyspnea for 6 months, who had the huge mass that almost completely occupied the esophageal lumen, was referred to our institution for the treatment.We diagnosed the mass as non-invasive tumor that has a stalk at the close to the esophageal orifice, by the CT image using air injection into esophageal lumen. We performed excision of the pedunclated huge mobile mass by esophagotomy via right thoracic approach with use of endoloop. Pathological examination showed a lipoma. CONCLUSION: In conclusion, an adequate preoperative evaluation to identify the correct origin of the stalk is mandatory for a successful treatment. In order to do the adequate preoperative evaluation and successful surgery, our diagnostic method of CT image can be effective.

Acute respiratory distress syndrome due to Strongyloides stercoralis infection in a patient with cervical cancer.

A 62-year-old woman complained of diarrhea and vomiting after receiving chemotherapy for cervical cancer in association with high doses of corticosteroids. Two months later, the patient developed acute respiratory distress syndrome, and numerous Strongyloides stercoralis parasites were found in the intrabronchial discharge. Ivermectin was administered daily until nematodes were no longer detected in the sputum, and the patient's condition was successfully rescued. Antibodies for human T-cell lymphotropic virus-1 (HTLV-1) were positive. HTLV-1 infection and the administration of corticosteroids are known risk factors for strongyloides hyperinfection syndrome. Therefore, physicians should consider this disease in the differential diagnosis of patients from endemic areas who present with gastrointestinal symptoms under these risk factors.

Increased expression of HBZ and Foxp3 mRNA in bronchoalveolar lavage cells taken from human T-lymphotropic virus type 1-associated lung disorder patients.

OBJECTIVE: Human T-lymphotropic virus type 1 (HTLV-I) causes adult T-cell leukemia/lymphoma (ATLL), and is associated with chronic inflammatory diseases, including inflammatory pulmonary diseases. HTLV-I bZIP factor (HBZ), which is expressed in all adult T-cell leukemia cells, plays a critical role in the development of lymphoma and systemic inflammation. HTLV-I is harbored by CD4(+) T cells that express forkhead box P3 (Foxp3), and HBZ interacts with Foxp3. This study investigated the chest computed tomography (CT) findings and expression of HBZ and Foxp3 in the bronchoalveolar lavage (BAL) cells from patients with HTLV-I-associated lung disorders. METHODS: CT scans obtained from 37 patients (10 men and 27 women, aged 37-77 years) with HTLV-I-associated lung disorders were retrospectively evaluated. The expression levels of HBZ and Foxp3 mRNA in BAL cells and the levels of inflammatory cytokines in the BAL fluid (BALF) from patients were compared with those in control subjects. RESULTS: CT scans frequently revealed a diffuse panbronchiolitis (DPB)-like pattern, along with a nonspecific interstitial pneumonia (NSIP) pattern. An analysis of the BALF revealed lymphocytosis and increased expression of HBZ mRNA in patients with HTLV-I-associated lung disorders. The expression of Foxp3 mRNA positively correlated with the percentages of lymphocytes present in the BALF. The inflammatory cytokine and IL-10 levels were significantly increased in the BALF from patients with HTLV-I-associated lung disorders. CONCLUSION: The NSIP pattern may be a manifestation of pulmonary involvement in HTLV-I-infected patients, as is the DPB-like pattern. HBZ and Foxp3 likely have a role in the development of lung inflammation.

Mechanisms involved in the extension of pulmonary Mycobacterium avium infection from the pulmonary focus to the regional lymph nodes.

PURPOSE: This study was designed to evaluate the mechanism of Mycobacterium avium extension in lung infection. STUDY DESIGN: Retrospective study. PARTICIPANTS: A 42-year-old man with acquired immune deficiency syndrome and immune reconstitution inflammatory syndrome. The patient developed mediastinal lymphadenopathy and a peripheral lesion in the right upper lobe within 2 weeks of starting highly active antiretroviral therapy. METHODS: Pulmonary tissue and lymph nodes were dissected under thoracoscopy and evaluated pathologically and immunohistochemically. RESULTS: Pulmonary pathologic examination revealed extensive granuloma formation throughout the acini. Mycobacterial antigens were found in the macrophages in the alveoli and in the alveolar septa. Some macrophages including mycobacterial antigens were surrounded by lymphatic endothelial cells in the interstitium. In addition, a proliferative granulomatous lesion was found under the intact epithelial layer of a bronchiole. Pathological examination of the lymph nodes revealed aggregated proliferative granulomas with few mycobacteria. Genetically closely related M. avium strains were isolated from both tissues. CONCLUSIONS: This study showed the mechanism involved in the progression of pulmonary M. avium infection from the pulmonary focus to the regional lymph nodes via the lymphatic vessels.

Influence of human T lymphotropic virus type I infection on the etiology of community-acquired pneumonia.

BACKGROUND: Human T lymphotropic virus type I (HTLV-I), the cause of human T cell leukemia, is associated with a high incidence of several other infectious diseases. However, the relationship between pulmonary infections and HTLV-I infection is still unclear. OBJECTIVE: A large-scale retrospective study was conducted on hospital inpatients to evaluate the relationship between community-acquired pneumonia (CAP) and HTLV-I infection. METHODS: The present study included 4,666 hospitalized patients during 1991-2007. Three hundred and thirteen of them were diagnosed as CAP. The presence of serum HTLV-I antibody was determined in all patients on admission. Prevalence of HTLV-I infection was analyzed between CAP patients and all inpatients. We also compared HTLV-I-positive CAP patients and HTLV-I-negative CAP patients for severity and manifestation of pneumonia. RESULTS: The prevalence of HTLV-I was higher in CAP patients than in all inpatients (18.9%: 13.7%, p=0.011). The rates of renal diseases and collagen vascular disorders were higher in the HTLV-I-positive CAP patients than in the HTLV-I-negative CAP patients. Multivariate analysis revealed that HTLV-I infection, gender, COPD and collagen vascular disorders were all independent risk factors for CAP. The severity indices of CAP, the PORT score and the CURB-65 score, were higher in the HTLV-I-positive patients than in the HTLV-I-negative patients. CONCLUSION: This study demonstrates that HTLV-I infection might be an independent risk factor for CAP and that HTLV-I-infected patients tend to demonstrate a relatively severe form of pneumonia.

Legionella pneumonia caused by Legionella pneumophila serogroup 2: second case report in Japan.

A 56-year-old man with a 3-day history of a chilly sensation and general fatigue presented to a hospital in his neighborhood. He was diagnosed as having pneumonia and immediately treated with intravenous ceftriaxone sodium, but his respiratory condition deteriorated and he developed symptoms of restlessness. Although Legionella urinary antigen detection tests were negative, his clinical course suggested Legionella pneumonia. After his treatment was changed to intravenous ciprofloxacin and oral clarithromycin, his general condition gradually improved. Later, Legionella pneumophila serogroup 2 was isolated from a bronchoalveolar lavage specimen. This was considered to be the causative organism. In our literature search, this was only the second case of Legionella pneumonia caused by Legionella pneumophila serogroup 2 in Japan.

[A-DROP system might result in underestimation of severity of cases with Legionnaires' disease].

Fifteen cases of legionella pneumonia experienced in our department or associated hospital were included in this study. Each case was classified with the A-DROP system of the Japanese Respiratory Society Guidelines, and guidelines from the Infectious Diseases Society of America (IDSA) (1998) and then we compared the severity of the cases of pneumonia. Although 10 cases were classified as intermediate, 3 as severe, and 2 as extremely severe with the A-DROP system, most cases were classified as severe according to the IDSA guidelines. Among 5 fatal cases, three were ranked as intermediate with the A-DROP system. However all the fatal cases were ranked as severe in the IDSA guidelines. This study suggested that the A-DROP system might underestimate the severity of pneumonia in cases with legionella pneumonia.

[A case of multisystem Langerhans' cell histiocytosis with lung deterioration over 15 years].

We report a case of multisystem Langerhans cell histiocytosis (LCH) with lung, bone and pituitary involvement. A 20-year-old man developed thirst, polydipsia and polyuria in 1983. He had right femur pain from 1988 and osseous LCH was diagnosed based on the operated specimen in 1989. From July 1990, he had right chest pain on coughing and dyspnea and was admitted in November 1990. LCH involving the lungs was diagnosed by CT images and diabetes insipidus was also detected. Steroid therapy was started from 1991, but he discontinued it in 1998. Though he stopped smoking, his clinical symptoms worsened and he experienced bilateral pneumothoraces in 2002 and since then he has been receiving home oxygen therapy. Pulmonary LCH is thought to have a good prognosis, but in recent studies, its survival rate appears low. We report a case of multisystem LCH with lung deterioration over about 15 years.