Diagnosis and management of intercostal intramuscular hemangioma: an updated review.

BACKGROUND: Intramuscular hemangioma (IMH) is an uncommon type of hemangioma, and primary IMH of the intercostal muscle is even rarer. Only a few reports describe IMH of the intercostal muscle, and there are no review articles on this topic. We report our experience with a younger female patient, who underwent video-assisted thoracic surgery with tumor resection and review the previous literatures of intercostal IMH. CASE PRESENTATION: An asymptomatic 17-year-old woman showed a 29-mm, homogeneous, intrathoracic nodule in the left chest wall, attached to the second and third ribs on computed tomography. We performed exploratory thoracoscopic surgery and the tumor was excised without surrounding rib resection. Histopathologic examination of the surgical specimen revealed proliferation of small blood vessels within the surrounding striated muscle, leading to the diagnosis of intercostal IMH. The surgical margin was negative. The patient's postoperative course was uneventful, and there has been no evidence of recurrence for more than 18 months after surgery. CONCLUSIONS: We describe a case of intercostal IMH, who received tumor resection with clear excision margin without surrounding rib resection. Preoperative diagnosis is challenging due to its rarity, but intercostal IMH should be recalled as a differential diagnosis of chest wall tumor. Tumor excision without surrounding rib resection is acceptable for intercostal IMH, when there is a good possibility of achieving negative surgical margin.

Venous Stent Placement for Malignant Vena Cava Syndrome in a Patient with Liposarcoma.

BACKGROUND Reports of venous stenting for inferior vena cava (IVC) syndrome (IVCS) due to sarcoma are limited, and the treatment's efficacy and safety are not clear. CASE REPORT A 36-year-old woman with myxoid liposarcoma was admitted to the Department of Respiratory Medicine for treatment of bilateral lower-leg edema and to be evaluated for acute liver dysfunction. She was 13 years old when she was diagnosed with myxoid liposarcoma. Over the next 18 years, she had 4 tumor resections and 1 round of radiation therapy. She had been on chemotherapy for 4 years and then pazopanib at the age of 35. The edema did not improve after admission despite treatment with diuretics. Computed tomography revealed a huge liposarcoma occupying the right thoracic cavity and a compressed IVC, which caused the edema. Although doxorubicin was administered as fifth-line treatment, there was no response. Since there was no additional chemotherapy regimen, her prognosis was considered to be less than 6 months. She could not be discharged to her home since she was unable to walk due to the edema; therefore, IVC stenting was performed to improve her dysmotility. After IVC stenting, the lower-leg edema improved without any adverse events, enabling her to walk and eventually return home. CONCLUSIONS In patients with IVCS caused by rare malignancies such as myxoid liposarcoma, an IVC stent can be safely implanted and can help to alleviate symptoms. IVC stenting can improve symptoms and allow for home care, resulting in improved quality of life.

Use of Indocyanine Green Fluorescence Imaging in Thoracic and Esophageal Surgery.

BACKGROUND: Fluorescence imaging using indocyanine green in thoracic and esophageal surgery is gaining popularity because of the potential to facilitate surgical planning, to stage disease, and to reduce postoperative complications. To optimize use of fluorescence imaging in thoracic and esophageal surgery, an expert panel sought to establish a set of recommendations at a consensus meeting. METHODS: The panel included 12 experts in thoracic and upper gastrointestinal surgery from Asia-Pacific countries. Before meeting, 7 focus areas were defined: intersegmental plane identification for sublobar resections; pulmonary nodule localization; lung tumor detection; bullous lesion detection; lymphatic mapping of lung tumors; evaluation of gastric conduit perfusion; and lymphatic mapping in esophageal surgical procedures. A literature search of the PubMed database was conducted using keywords indocyanine green, fluorescence, thoracic, surgery, and esophagectomy. At the meeting, panelists addressed each focus area by discussing the most relevant evidence and their clinical experiences. Consensus statements were derived from the proceedings, followed by further discussions, revisions, finalization, and unanimous agreement. Each statement was assigned a level of evidence and a grade of recommendation. RESULTS: A total of 9 consensus recommendations were established. Identification of the intersegmental plane for sublobar resections, localization of pulmonary nodules, lymphatic mapping in lung tumors, and assessment of gastric conduit perfusion were applications of fluorescence imaging that have the most robust current evidence. CONCLUSIONS: Based on best available evidence and expert opinions, these consensus recommendations may facilitate thoracic and esophageal surgery using fluorescence imaging.

Assessing the development status of intraoperative fluorescence imaging for anatomy visualisation, using the IDEAL framework.

Objectives: Intraoperative fluorescence imaging is currently used in a variety of surgical fields for four main purposes: visualising anatomy, assessing tissue perfusion, identifying/localising cancer and mapping lymphatic systems. To establish evidence-based guidance for research and practice, understanding the state of research on fluorescence imaging in different surgical fields is needed. We evaluated the evidence on fluorescence imaging used to visualise anatomical structures using the IDEAL framework, a framework designed to describe the stages of innovation in surgery and other interventional procedures. Design: IDEAL staging based on a thorough literature review. Setting: All publications on intraoperative fluorescence imaging for visualising anatomical structures reported in PubMed through 2020 were identified for five surgical procedures: cholangiography, hepatic segmentation, lung segmentation, ureterography and parathyroid identification. Main outcome measures: The IDEAL stage of research evidence was determined for each of the five procedures using a previously described approach. Results: 225 articles (8427 cases) were selected for analysis. Current status of research evidence on fluorescence imaging was rated IDEAL stage 2a for ureterography and lung segmentation, IDEAL 2b for hepatic segmentation and IDEAL stage 3 for cholangiography and parathyroid identification. Enhanced tissue identification rates using fluorescence imaging relative to conventional white-light imaging have been documented for all five procedures by comparative studies including randomised controlled trials for cholangiography and parathyroid identification. Advantages of anatomy visualisation with fluorescence imaging for improving short-term and long-term postoperative outcomes also were demonstrated, especially for hepatobiliary surgery and (para)thyroidectomy. No adverse reactions associated with fluorescent agents were reported. Conclusions: Intraoperative fluorescence imaging can be used safely to enhance the identification of anatomical structures, which may lead to improved postoperative outcomes. Overviewing current research knowledge using the IDEAL framework aids in designing further studies to develop fluorescence imaging techniques into an essential intraoperative navigation tool in each surgical field.

Membranous HEG1 expression is a useful marker in the differential diagnosis of epithelioid and biphasic malignant mesothelioma versus carcinomas.

Sialylated HEG1 has been reported as a highly specific and sensitive mesothelioma marker but a comprehensive evaluation of its expression in carcinomas in different organs, various sarcomas and reactive mesothelial proliferations has not been reported. The aim of this study was to evaluate the clinical applicability of HEG1 as a marker in the diagnosis of mesothelioma. HEG1 immunoreactivity was evaluated in whole sections of 122 mesotheliomas, 75 pulmonary carcinomas, 55 other carcinomas, 16 mesenchymal tumors, and 24 reactive mesothelial proliferations and in tissue microarrays containing 70 epithelioid (EM), 36 biphasic (BM), and 2 sarcomatoid mesotheliomas (SM). In whole sections and tissue microarrays, respectively, membranous HEG1 was expressed in 93.0% and 85.5% of EM, 81.3% and 69.4% of BM, 0% and 0% of SM. HEG1 was not expressed in pulmonary adenocarcinomas. HEG1 was expressed as cytoplasmic immunoreactivity in pulmonary squamous cell carcinomas (21.7%). Membranous HEG1 staining was seen in ovarian carcinomas (66.7%), thyroid carcinomas (100%), reactive conditions (16.7%), and mesenchymal tumors (18.8%). The sensitivity of membranous HEG1 expression to distinguish EM/BM from all carcinomas was 88.8%. The specificity for the differential diagnosis between EM/BM and all carcinomas and pulmonary carcinomas was 92.3% and 98.7%, respectively.

The efficacy of transbronchial indocyanine green instillation for fluorescent-guided wedge resection.

OBJECTIVES: The purpose of this study was to investigate the feasibility of lung wedge resection by combining 3-dimensional (3D) image analysis with transbronchial indocyanine green (ICG) instillation, in order to delineate the intended area for resection. METHODS: From December 2017 to July 2020, 28 patients undergoing wedge resection (17 primary lung cancers, 11 metastatic lung tumours) were enrolled, and fluorescence-guided wedge resection was attempted. Virtual sublobar resections were created preoperatively for each patient using a 3D Image Analyzer. Surgical margins were measured in each sublobar resection simulation in order to select the most optimal surgical resection area. After transbronchial instillation of ICG, near-infrared thoracoscopic visualization allowed matching of the intended area for resection to the virtual sublobar resection area. To investigate the effectiveness of ICG instillation, the clarity of the ICG-florescent border was evaluated, and the distance from the true tumour to the surgical margins was compared to that of simulation. RESULTS: Mean tumour diameter was 12.4 ± 4.3 mm. The entire targeted tumour was included in resected specimens of all patients (100% success rate). The shortest distances to the surgical margin via 3D simulation and by actual measurement of the specimen were11.4 ± 5.4 and 12.2 ± 4.1 mm, respectively (P = 0.285) and were well correlated (R2 = 0.437). While all specimens had negative malignant cells at the surgical margins, one loco-regional recurrence was observed secondary to the dissemination of neuroendocrine carcinoma. CONCLUSIONS: ICG-guided lung wedge resection after transbronchial ICG instillation and preoperative 3D image analysis allow for adequate negative surgical margins, providing decreased risk of local recurrence.

HEG1, BAP1, and MTAP are useful in cytologic diagnosis of malignant mesothelioma with effusion.

BACKGROUND: The specificity and sensitivity of HEG1 for malignant mesothelioma (MM) is high. The use of BAP1/MTAP immunohistochemistry (IHC) is recommended to separate benign and malignant mesothelial proliferations. We determined how ancillary techniques can be used for the cytological diagnosis of MM with effusion. METHODS: Cell blocks from effusions from cases with MM, reactive mesothelial cells (RMCs), and carcinomas were analyzed by IHC with HEG1, BAP1, and MTAP and with homozygous deletion (HD) of CDKN2A by fluorescence in situ hybridization. Staining scores were calculated for IHC by adding the number of categories for the staining intensity and the staining extension. RESULTS: HEG1 was positive in all (41/41) MMs, but negative in carcinomas, except for ovarian carcinomas. Overall 76.9% (20/26) of RMCs and 28.6% (6/21) of ovarian carcinomas expressed HEG1. BAP1 loss was found in 71.1% of MMs, but none was found in RMCs. MTAP loss was found in 76.2% of MMs, but none was found in RMCs. 73.9% of MMs harbored HD of CDKN2A. There was concordance between loss of MTAP and HD of CDKN2A in 95% of MMs. CONCLUSION: HEG1 is a good marker for mesothelial differentiation in effusion cytology. HD of CDKN2A is frequently observed in cell blocks from effusions of MMs, and MTAP IHC may act as a surrogate for HD of CDKN2A. Cell block analysis is recommended for effusions of unknown origins with the following methods: IHC with HEG1 and claudin 4 to validate the mesothelial origin, followed by BAP1 and MTAP IHC to confirm malignancy.

PD-L1, FGFR1, PIK3CA, PTEN, and p16 expression in pulmonary emphysema and chronic obstructive pulmonary disease with resected lung squamous cell carcinoma.

BACKGROUND: Emphysema and chronic obstructive pulmonary disease (COPD) are well known independent risk factors for lung cancer. However, the developmental mechanisms between emphysema/COPD and lung cancer remain unknown. The purpose of this study was to evaluate PD-L1, FGFR1, PIK3CA, PTEN, and p16 expression in squamous cell carcinoma (SCC) associated with emphysema/COPD. METHODS: A total of 59 patients with squamous cell lung carcinoma (SCC) resected between 2008 and 2012 were retrospectively reviewed. Emphysema was assessed according to the Goddard score. Total severity was divided into none-mild (0-7), moderate (8-15), and severe (≥ 16). Local severity around the existing tumor was divided into no emphysema (0) and presence of emphysema (1-4). COPD severity was based on the Global Initiative for Chronic Obstructive Lung Disease (GOLD) criteria. PD-L1, FGFR1, PIK3CA, PTEN, and p16 expression were evaluated by immunohistochemistry (IHC). Expression level was classified as tumor cells (TC) 3 (≥ 50%), TC2 (5-49%), TC1 (1-4%), or TC0 (< 1%), and as tumor-infiltrating immune cells (IC) 3 (≥ 50%), IC2 (5-49%), IC1 (1-4%), or IC0 (< 1%) for PD-L1. Expression level was compared between none-mild/moderate-severe total emphysema, no/presence of local emphysema, no COPD/COPD, and GOLD 1/GOLD 2, 3. RESULTS: PD-L1 expression was significantly correlated with severity of emphysema in TC0, 1, 2 vs. TC3 (P = 0.012). PD-L1 was significantly higher inversely in none-mild emphysema compared to moderate-severe (95% CI, 0.061-5.852, P = 0.045). There were no other significant associations between PD-L1, FGFR1, PIK3CA, PTEN, and p16 expression and total/local severity of emphysema or presence of COPD/GOLD stage. CONCLUSIONS: PD-L1 expression in SCC was correlated with severity of emphysema in TC0, 1, 2 vs. TC3 and more frequent in none-mild emphysema than moderate-severe emphysema.

[Fluorescence Guided Sublobar Lung Resection by Using Three-dimensional Image Analysis and Transbronchial Instillation of Indocyanine Green].

BACKGROUND: The confirmation of an appropriate resection margin from the tumor is crucial for reducing the risk of local recurrence after sublobar resection for pulmonary malignancies. PATIENTS AND METHODS: From October 2014 to April 2018, 66 operative cases in 64 patients( primary lung cancer 42, metastatic lung tumor 21, benign disease 3) were enrolled. In lung cancer, active limited resection was done in 29 and passive limited resection was done in 13. Preoperatively, each patient created several virtual sublobar resections by using 3-dimensional (3D) volume analyzer. We measured the surgical margin in each simulated sublobar resection and selected the most appropriate procedure. Surgical resection matched with virtual sublobar resection was done by using an infrared thoracoscopy with transbronchial indocyanine green (ICG) instillation. In lung cancer, we compared surgical outcomes between ICG cases and 47 historical segmentectomy cases. RESULTS: The types of sublobar resection were subsegmental resection in 5, simple segmentectomy in 15, complex segmentectomy in 16 and extended segmentectomy in 22 and anatomical super deep wedge resection in 8. The shortest distance of surgical margin by simulation and an actual measurement were 20.8±11.1 mm and 22.6±8.3 mm, respectively( p=0.186). Postoperative recurrence was found in 8 cases (distant in 7 and mediastinal lymph node in 1). No locoregional recurrence was found in all cases. Postoperative recurrence was similar between the 2 groups in active and passive limited resection, respectively. CONCLUSION: ICG-guided sublobar resection by transbronchial ICG instillation is applicable to any type of sublobar resection and can control local recurrence of lung neoplasms.

Surgical outcome of pulmonary artery reconstruction using the expanded polytetrafluoroethylene patch in patients with lung cancer.

PURPOSE: Pulmonary artery reconstruction is sometimes utilized as an alternative to pneumonectomy in lung cancer surgery. We herein report our experience of pulmonary artery reconstruction using an expanded polytetrafluoroethylene (ePTFE) patch based on the surgical results and long-term outcome. METHODS: Clinical records of lung cancer patients who underwent patch plasty were reviewed retrospectively. RESULTS: Between 2003 and 2017, pulmonary artery patch plasty were performed in 21 patients [18 males, 3 females; mean age 65 (range 47-79) years]. Induction chemoradiotherapy was performed in three patients. Bronchoplasty was performed in five patients. The pathologic stages were stage I in 3 patients, stage II in 6 and stage III in 12. Pneumonectomy, lobectomy and segmentectomy were performed in 2, 18 and 1 patient, respectively. The left upper lobe was the most frequent origin of lung cancer (15 patients). There was no reconstruction-related morbidity or mortality. The overall survival rate at 5 years was 64.1% with a mean follow-up of 39.5 months, and the survival rates for N0-1 and N2-3 were 80.8% and 28.6%, respectively. CONCLUSION: Patch angioplasty using the ePTFE sheet is a reliable procedure in radical surgery for lung cancer.

Precise Anatomical Sublobar Resection Using a 3D Medical Image Analyzer and Fluorescence-Guided Surgery With Transbronchial Instillation of Indocyanine Green.

We developed a novel approach combined with 3D image analyzer and infrared thoracoscopy for pulmonary sublobar resection. The purpose of this study was to investigate the feasibility of this procedure. From October 2014 to April 2018, 65 cases were enrolled, and 58 cases were evaluated. For each case, several virtual sublobar resections were created by 3D image analyzer preoperatively. The surgical margin was measured in each simulated sublobar resection and the most appropriate procedure was selected. Surgical resection with matching virtual sublobar resection was performed using infrared thoracoscopy with transbronchial indocyanine green (ICG) instillation. We evaluated the border clarity of ICG fluorescence to investigate success of ICG injection and compared pre- and postoperative CTs to determine whether the correct area could be removed according to the simulation. We also compared short-term surgical outcomes between the ICG cases and historical segmentectomy cases by propensity score matching. The success rate of transbronchial ICG injections was 89.2% (58/65). These 58 patients were eligible for evaluation of our procedure. Sublobar resection included subsegmental resection (5), simple segmentectomy (15), complex segmentectomy (16), and extended segmentectomy (22). The shortest distances to the surgical margin by simulation and by actual measurement were 21.5 ± 11.2 mm and 23.5 ± 8.3, respectively (P = 0.190). Fifty-four of 58 cases underwent sublobar resection matched with the simulation (93.1% concordance rate). Operative results and short-term outcomes were similar between the 2 groups by propensity score matching. ICG-guided sublobar resection by transbronchial ICG instillation is feasible and applicable to any type of sublobar resection.

A surgical case of lung cancer with poor general status associated with parathyroid hormone-related protein.

Parathyroid hormone-related protein (PTHrP), which is released in the presence of malignant disease, is associated with hypercalcemia. Complete resection of the tumor in such patients is rarely performed because of their poor general condition. We herein report a case of lung cancer associated with PTHrP in a patient whose condition dramatically improved after surgery. We also review the literature on the benefits of various surgical options. Although only a few cases of complete resection in such patients have been reported, the mental and physical condition of the patients improved postoperatively and the median survival time was longer than 12 months. A poor general status is frequently considered a contraindication for surgery, even in a palliative setting; however, we conclude that resection of lung cancer may lead to improved symptom control and survival when the patient's condition is induced by hypercalcemia secondary to PTHrP secretion from the tumor.

Safety and efficacy following 10-years of overnight orthokeratology for myopia control.

PURPOSE: To compare rates of myopia progression and adverse events between orthokeratology (OK) and soft contact lens (SCL) wearers over a 10-year period in schoolchildren. METHODS: Medical records of consecutive patients (≤16 years of age at baseline) who started OK for myopia correction and continued the treatment for 10 years were retrospectively reviewed. For the control group, patients who started using soft contact lenses (SCLs) for myopia correction and continued to use them for 10 years were also reviewed. Clinical data, including sex, age, manifest refraction, visual acuity, prescription lens power, and adverse events during the 10-year period, were recorded. Estimated myopia progression was calculated as the sum of 'changes in prescription lens power during 10 years' and 'residual refractive errors at the 10-year visit,' and was compared between groups. We also compared the incidence of adverse events between groups over the 10-year study period. RESULTS: A total of 104 eyes of 53 patients who underwent OK treatment and 78 eyes of 39 patients who wore SCLs fulfilled the criteria. The estimated myopia progression over the 10-year period found in the OK and SCL groups were -1.26 ± 0.98 and -1.79 ± 1.24 days, respectively; this difference was statistically significant (p = 0.001). Additionally, lower myopia progression was found in the OK in comparison to the SCL group at all baseline ages (p = 0.003 to p = 0.049) except at 16 years old (p = 0.41). No significant difference was found in the number of adverse events found between the OK (119) and SCL (103) groups (p = 0.72). CONCLUSIONS: The results of this study supports the long-term efficacy and safety of OK lens wear in reducing myopia progression in schoolchildren.

Possible relationship between fibrosis of IgG4-related thymitis and the profibrotic cytokines, transforming growth factor beta 1, interleukin 1 beta and interferon gamma: a case report.

BACKGROUND: IgG4-related disease often forms a mass and the affected lesion is clinically removed because the mass cannot be differentiated from a neoplasm. Affected lesions commonly occur in the pancreas, hepatobiliary tract, kidney, and retroperitoneum. However, the lesion rarely occurs in the thymus. A histological worldwide consensus of IgG4-related disease proposed that pathological diagnosis of IgG4-related disease should meet more than two of three major features: 1) dense lymphoplasmacytic infiltration with greater than 40% IgG4+/IgG+ plasma cells, 2) storiform fibrosis; and 3) obliterative phlebitis. Currently, fibrosis of IgG4-related disease is thought to be induced by profibrotic cytokines such as transforming growth factor beta 1 (TGFB1), interleukin 1 beta (IL1B) and interferon gamma (IFNG), which are secreted by regulatory T cells (Tregs) and CD4-positive cytotoxic T cells. However, it is unclear whether profibrotic cytokines are associated with the fibrosis seen in IgG4-related thymitis. Here we examined whether cytokines in the mass were increased compared with those in the surrounding thymus, and whether Tregs were present in the mass, using reverse transcription absolute quantitative polymerase chain reaction (RT-ab-qPCR) and immunohistochemistry. CASE PRESENTATION: A 70-year-old Japanese man contracted IgG4-letated thymitis. Histological and immunohistochemical analyses demonstrated his mass had massive fibrosis with a focally storiform pattern and lymphoplasmacytic infiltration with 40% IgG4+/IgG+ plasma cells, but not obliterative phlebitis. The mass was surrounded by atrophic thymus. We diagnosed the mass as IgG4-related thymitis. Immunohistochemically, Tregs were scattered throughout the mass. RT-ab-qPCR showed that messenger RNA expressions of TGFB1, IL1B and IFNG in the mass were 270-, 158- and 5.5- fold higher than in the surrounding thymus. His serum IgG4 level after surgery was within the normal range (83.4 mg/dl soon after surgery, 89.3 mg/dl 2 weeks after surgery). CONCLUSIONS: Our results suggested the profibrotic cytokines TGFB1, IL1B and IFNG induce fibrosis and that Tregs might produce some of these cytokines in IgG4-related thymitis as well as in the other affected lesions of IgG4-related disease.

Evaluating vertebral artery dominancy before T4 lung cancer surgery requiring subclavian artery reconstruction.

PURPOSES: To evaluate vertebral artery (VA) dominancy and the risk of brain infarction in T4 lung cancer patients with tumor invasion into the subclavian artery. METHODS: We reconstructed the subclavian artery in 10 patients with T4 non-small cell lung cancer. The histological stages were IIIA in eight patients and IIIB in two patients. We evaluated the VA dominancy by performing a four-vessel study preoperatively and investigated the relationship between the methods of VA treatment and postoperative brain complications, retrospectively. RESULTS: Seven patients had a superior sulcus tumor (SST) and three had direct invasion into the mediastinum. Based on the tumor location, a transmanublial approach was used in five patients and a posterolateral hook incision was used in the other five. All subclavian artery (SA) reconstructions were done using an artificial woven graft. Preoperative angiography of the VA revealed poor development of the contralateral side in two patients. One of these patients suffered a severe brain infarction on postoperative day 2, which proved fatal. In the other patient, the VA was connected to the left SA graft by a side-to-end anastomosis and there was no postoperative brain complication. CONCLUSIONS: Preoperative SA and VA angiography is mandatory for identifying the need for VA reconstruction in lung cancer patients with major arterial invasion.

The efficacy of transbronchial needle aspiration with endobronchial ultrasonography using a guide sheath for peripheral pulmonary lesions suspected to be lung cancer.

BACKGROUND: The efficacy of transbronchial needle aspiration (TBNA) with endobronchial ultrasonography using a guide sheath (EBUS-GS) for cases of peripheral pulmonary lesions (PPLs) has not been well established. The purpose of this study was to evaluate the efficacy of TBNA with EBUS-GS for PPLs. METHODS: We evaluated 130 patients suspected to have lung cancer who underwent transbronchial brushing (brushing), transbronchial biopsy (TBB), and TBNA with EBUS-GS. The pathological diagnostic yields of TBNA were compared to that of TBB and brushing. The histological diagnosis of TBNA was compared to that of surgical specimens. The results of epidermal growth factor receptor (EGFR) gene mutation in TBNA samples were compared to that in TBB or surgical specimens. RESULTS: The diagnostic yields of this study were 62.9% for brushing, 80.0% for TBB, and 77.1% for TBNA. Histological diagnosis was 84.8% for TBB and/or TBNA and pathological diagnosis was 86.7% for all the procedures. TBB and TBNA had significance higher than brushing (p < 0.05). TBB and TBNA had a tendency of higher diagnostic yields than brushing if EBUS probe was adjacent to PPLs (p = 0.058). Histological evaluations were obtained from TBNA specimens from 50 of 105 patients (47.6%) and these were identical to those of surgical specimens from 29 of 32 patients (90.6%). The results of EGFR gene mutation in TBNA specimens were identical to the same tissue obtained by surgery or TBB. CONCLUSIONS: TBNA with EBUS-GS for PPLs was a useful tool for accurate diagnosis and EGFR gene mutation analysis. This method may improve diagnostic accuracy and be useful for molecular testing. This study was approved by the institutional review board (Date of approval: May 27, 2013, approval number: 2816) of Tokyo Women's Medical University Hospital.

Intravenous Immunoglobulin Monotherapy for Granulomatous Lymphocytic Interstitial Lung Disease in Common Variable Immunodeficiency.

Common variable immunodeficiency (CVID) is a heterogeneous subset of immunodeficiency disorders. Recurrent bacterial infection is the main feature of CVID, but various non-infectious complications can occur. A 42-year-old woman presented with cough and abnormal chest X-ray shadows. Laboratory tests showed remarkable hypogammaglobulinemia. Computed tomography revealed multiple consolidation and nodules on the bilateral lung fields, systemic lymphadenopathy, and splenomegaly. A surgical lung biopsy specimen provided the final diagnosis of lymphoproliferative disease in CVID, which was grouped under the term granulomatous lymphocytic interstitial lung disease. Interestingly, the lung lesions of this case resolved immediately after the initiation of intravenous immunoglobulin monotherapy.

A case of respiratory infection possibly caused by Mycobacterium triviale: Current problems on diagnostic and therapeutic strategies.

Mycobacterium triviale is a subspecies of the Mycobacterium terrae complex, which rarely causes disease in humans. We encountered a case of respiratory infection, possibly caused by M. triviale, which was successfully treated by levofloxacin and clarithromycin. Although DNA-DNA hybridization identified M. triviale in one of three samples, clinical validations convinced us that it was the pathogen. 16s ribosomal RNA sequencing would have been reliable and ideal to perform in this case, although it is not covered by the insurance system in Japan. Nevertheless, this experience remains to be instructive because the clinical course, guidelines on the diagnosis, and therapeutic strategies for respiratory infections caused by M. triviale are not well-known or have not been established. Awareness of the possibility of respiratory infections caused by M. triviale and further collection and analysis of its predisposing conditions are essential.

Rosai-Dorfman Disease of the Lung Overlapping with IgG4-related Disease: The Difficulty in Its Differential Diagnosis.

We herein report a case of Rosai-Dorfman disease (RDD) overlapping with IgG4-related disease (IgG4-RD), which presented as diffuse interstitial lung disease with a perilymphatic pattern, followed by submandibular gland and eyelid swelling. The pathological findings of the submandibular gland biopsy specimen were indicative of IgG4-RD alone. We diagnosed the patient with RDD with overlapping IgG4-RD. However, the optimal method for differentiating between these two entities is still controversial. It is important that clinicians are aware that RDD should be included in the differential diagnoses of diffuse interstitial lung disease with a perilymphatic pattern and that RDD can overlap with IgG4-RD.