Spontaneous Pneumomediastinum in an Adolescent Soccer Player.

Spontaneous pneumomediastinum (SPM) is an uncommon and usually benign self-limiting clinical disorder found in young people, often without apparent precipitating factors or diseases. A pressure gradient exists between the peripheral pulmonary alveoli and the hilum, and increased intra-alveolar pressure causes rupture of the terminal alveoli. We present the case of a 15-year-old male soccer player who presented with a complaint of anterior chest pain and dysphagia after stopping the strong ball with his chest. His symptom gradually progressed over hours. We can make the diagnosis of SPM using by chest X-ray and computed tomography (CT) scanning. His symptoms were gradually resolved over the course of approximately one week with no exercise and careful observation. We believe that our case provides very useful information to alert clinicians and coaches regarding this rare disease that may occur in anyone including adolescent soccer players.

Novel clinical scoring system to identify patients with pneumothorax with suspicion for Birt-Hogg-Dubé syndrome.

BACKGROUND AND OBJECTIVE: Birt-Hogg-Dubé syndrome (BHDS) is a rare hereditary disease that presents with multiple lung cysts and pneumothorax (PTX). Although some reports propose that findings from chest computed tomography enable one to distinguish BHDS from primary spontaneous pneumothorax (PSP), it is still unclear whether clinical features are useful for identifying patients with suspicion of BHDS from those with PTX. METHODS: We retrospectively reviewed the medical records of patients with PTX who underwent video-assisted thoracoscopic surgery at Nissan Tamagawa Hospital from January 2012 to December 2015. RESULTS: We identified a total of 1141 patients with PTX, including 54 with BHDS and 517 with PSP. Among them, logistic regression analysis segregated five features that were significantly associated with BHDS: familial history of PTX, past history of bilateral PTX, age at the first episode of PTX (≥25 years old (y.o.)), body mass index (≥18.5) and gender (female). We assigned scores of 3, 3, 2, 2 and 1 to the five features, respectively, to establish a system with a calculated score from 0 to 11. The cut-off value of a calculated score ≥ 4 yielded the highest sensitivity of 93% and specificity of 86%. Receiver operating characteristic (ROC) analysis showed the area under the curve reflecting an accuracy of this diagnostic test as 0.953. CONCLUSION: BHDS has several clinical features distinct from PSP. Our scoring system consists of only five clinical variables that are easily evaluated and efficiently separate BHDS patients from those who have PTX without relying on an imaging study. Further prospective study is needed to confirm our findings.

Underweight and hypoalbuminemia as risk indicators for mortality among psychiatric patients with medical comorbidities.

AIM: Medical comorbidities are a major cause of death among patients with mental illness. The purpose of this study was to clarify the risk factors for mortality among psychiatric patients with medical comorbidities. METHODS: We retrospectively reviewed the clinical files of patients transferred to Tokyo Metropolitan Matsuzawa Hospital from a psychiatric hospital to treat medical comorbidities during the 3-year period from January 2014 to December 2016. We analyzed the clinical differences between the expired and alive patients. RESULTS: Of the 287 patients included, 29 (10.1%) had expired at the time of hospital discharge, while 258 (89.9%) were living. A multivariable analysis to determine the prognostic factors related to mortality from medical comorbidities showed that body mass index <18.5 had the highest odds ratio among the predictive factors (5.1; 95% confidence interval, 1.5-17.1; P < 0.05), followed by a serum albumin level < 3.0 mg/dL (3.0; 95% confidence interval, 1.1-8.1; P < 0.05). CONCLUSION: We found that underweight and hypoalbuminemia were risk factors for mortality among psychiatric patients with medical comorbidities. Physicians at psychiatric hospitals should consider transferring patients with medical comorbidities to a general medical hospital in the presence of underweight and/or hypoalbuminemia.

Immunomodulatory effects of peplomycin on immunosuppressive and cytotoxic cells in the lesional skin of cutaneous squamous cell carcinoma.

BACKGROUND: Continuous intra-arterial administration of peplomycin (PEP) through a tumor-feeding artery using an intravascular indwelling catheter is one of the best treatments for cutaneous squamous cell carcinoma (SCC) on cosmetic areas. Although this reagent is useful for the treatment of SCC, its immunomodulatory effect on the tumor microenvironment is still unknown. OBJECTIVE/METHODS: In this study, we investigated the immunomodulatory effects of PEP on the tumor-infiltrating regulatory T cells and tumor-associated macrophages as well as CD8(+)TIA-1(+) cytotoxic T cells in the lesional skin of 5 patients with SCC on the lips. RESULTS: Our data suggest that, in addition to the direct antitumor effects, PEP decreased immunosuppressive cells and increased cytotoxic T lymphocytes at the tumor sites, which might maintain antitumor immune response against SCC.

Clinical characteristics of catamenial and non-catamenial thoracic endometriosis-related pneumothorax.

BACKGROUND AND OBJECTIVE: A major pathogenic factor for catamenial pneumothorax is thoracic endometriosis. However, thoracic endometriosis-related pneumothorax (TERP) can develop as either catamenial or non-catamenial pneumothorax (CP). Therefore, the aim of this study was to elucidate the clinical differences between catamenial and non-catamenial TERP. METHODS: The clinical and pathological data in female patients who underwent video-assisted thoracoscopic surgery at the Pneumothorax Research Center during an 8-year period were retrospectively reviewed. This study included 150 female patients with surgico-pathologically confirmed TERP. The subjects were divided into two groups, those having all of the pneumothorax episodes in the catamenial period (CP group) and those who did not (non-CP group). We compared the clinical characteristics and surgico-pathological findings between these two groups. RESULTS: Of the 150 TERP patients, 55 (36.7%) were classified in the CP group, and 95 (63.3%) in the non-CP group. In regard to the locations of endometriosis, all TERP patients had diaphragmatic endometriosis, while pleural implantation was recognized in 34 of the 55 (61.8%) patients in the CP group and 42 of the 95 (44.2%) patients in the non-CP group (P < 0.05). CONCLUSIONS: A significant difference in the proportion of patients with pleural endometriosis was observed between catamenial and non-catamenial TERP. The ectopic sites of the endometriosis may be responsible for the timing of the pneumothorax episodes.

Eradication of hepatitis C virus could improve immunological status and pyoderma gangrenosum-like lesions.

Hepatitis C virus (HCV) can affect immune cells and induce various kinds of immune-related diseases including pyoderma gangrenosum. We experienced a difficult-to-treat case of pyoderma gangrenosum-like lesions in a patient with HCV infection. The patient was treated with pegylated interferon (PEG IFN)-α-2b and ribavirin (RBV) therapy and achieved a sustained virological response. Before the eradication of HCV, the frequency of T-helper 17 cells was remarkably high in comparison to chronic hepatitis C patients without extrahepatic immune-related diseases. Moreover, we could detect negative and positive strand-specific HCV RNA in the CD19(+) B lymphocytes and CD4(+) T lymphocytes. However, after the eradication of HCV, the immunological status became normal and the pyoderma gangrenosum-like lesions became stable without immunosuppressive therapy. Here, we report a sequential immunological analysis during PEG IFN/RBV therapy and the beneficial effect of HCV eradication in difficult-to-treat pyoderma gangrenosum-like lesions.

Thoracic endometriosis-related pneumothorax distinguished from primary spontaneous pneumothorax in females.

PURPOSE: Thoracic endometriosis-related pneumothorax (TERP) is a secondary condition specific for females, but in a clinical setting, TERP often is difficult to distinguish from primary spontaneous pneumothorax (PSP) based on a relationship between the dates of pneumothorax and menstruation. The purpose of this study was to clarify the clinical features of TERP compared with PSP. METHODS: We retrospectively reviewed the clinical and histopathological files of female patients with pneumothorax who underwent video-assisted thoracoscopic surgery in the Pneumothorax Research Center during the 6-year period from January 2005 to December 2010. We analyzed the clinical differences between TERP and PSP. RESULTS: The study included a total of 393 female patients with spontaneous pneumothorax, of whom 92 (23.4 %) were diagnosed as having TERP and 33.6 % (132/393) as having PSP. We identified four factors (right-sided pneumothorax, history of pelvic endometriosis, age ≥31 years, and no smoking history) that were statistically significant for predicting TERP and assigned 6, 5, 4, and 3 points, respectively, to establish a scoring system with a calculated score from 0 to 18. The cutoff values of a calculated score ≥12 yielded the highest positive predictive value (86 %; 95 % confidence interval (CI) 81.5-90.5 %) for TERP and negative predictive value (95.2 %; 95 % CI 92.3-98 %) for PSP. CONCLUSIONS: TERP has several distinct clinical features from PSP. Our scoring system consists of only four clinical variables that are easily obtainable and enables us to suspect TERP in female patients with pneumothorax.

Risk for re-expansion pulmonary edema following spontaneous pneumothorax.

PURPOSE: Re-expansion pulmonary edema is an uncommon condition that occurs when a collapsed lung is expanded. The aim of the present study was to investigate the incidence and risk factors associated with re-expansion pulmonary edema which may occur as a complication when carrying out treatment for spontaneous pneumothorax. METHODS: A total of 462 patients with spontaneous pneumothorax treated with chest tube drainage in inpatient settings at the Nissan Tamagawa Hospital during the 6-year period between January 2007 and December 2012 were retrospectively evaluated. The data were analyzed to identify any clinical differences between the patients with and without re-expansion pulmonary edema. RESULTS: Re-expansion pulmonary edema occurred on 30 (6.5 %) of the 462 patients. The duration of lung collapse in the patients with re-expansion pulmonary edema was longer than that observed in the patients without re-expansion pulmonary edema. (7.7 ± 9.1 and 2.4 ± 4.6 days). This difference was statistically significant (P < 0.0001). The extent of lung collapse in the patients with re-expansion pulmonary edema was more severe than that observed in the patients without re-expansion pulmonary edema. This difference was also statistically significant (P = 0.004). CONCLUSIONS: The results suggest that treating spontaneous pneumothorax using chest tube drainage requires careful consideration in view of the relatively high incidence of re-expansion pulmonary edema, especially in cases associated with long periods of lung collapse or large spontaneous pneumothoraxes.

Comparison of immunosuppressive and cytotoxic cells in angiosarcoma: development of a possible supportive therapy for angiosarcoma.

An imbalance of immunosuppressive and cytotoxic cells plays an important role in inhibiting the anti-tumor immune response of the tumor-bearing host. The purpose of this study was to elucidate the involvement of immunosuppressive cells, such as regulatory T cells and CD163+ M2 macrophages as well as cytotoxic cells, such as granulysin-bearing cells and TIA-1+ cells in cutaneous angiosarcoma (AS) by immunohistochemical staining. In addition we evaluated the potencies of bisphosphonate, which was previously reported to suppress the expression of matrix metalloproteinase 9 (MMP-9), as a supportive therapy for AS together with docetaxel in 6 cases of cutaneous AS. These findings suggest that a high number of immunosuppressive cells might be related to the prognosis of AS, and that a combination of docetaxel with bisphosphonate risedronate sodium might be effective for MMP-9-expressing AS.

Immunohistochemical analysis of thoracic endometriosis.

Thoracic endometriosis is a rare disease responsible for catamenial pneumothorax. The immunohistochemical features of thoracic endometriosis are not well understood. An immunohistochemical examination of 84 diaphragmatic specimens of catamenial pneumothorax using antibodies against estrogen receptor (ER), progesterone receptor (PgR), CD10 and smooth muscle actin (SMA) was conducted. The endometrial tissue was small, and focally located around the chasm of the tendon on the side of the thoracic cavity. Endometrial stroma were detected in 84/84 (100%) of the specimens, endometrial glands were detected in 21/84 (25%) and smooth muscle was detected in 1/84 (1.2%). The endometrial stroma exhibited positive staining for ER in 74/84 (88.1%) of the specimens, PgR in 84/84 (100%), CD10 in 74/84 (88.1%) and SMA in 46/84 (54.8%). Because thoracic endometriosis is small in size, and only 25% of the resected tissue specimens were accompanied with the endometrial gland, an immunohistochemical analysis can be useful for their detection. The fact that over half of the thoracic endometrial stroma showed positive staining for SMA, and the existence of thoracic endometriosis accompanied by smooth muscle, indicated that some part of the thoracic endometriosis may have the ability to differentiate into smooth muscle, although further studies are needed to confirm this hypothesis.

Arthroscopic All-Suture Anchor Technique for Unstable Ramp Lesions with Medial Meniscal Defects.

A ramp lesion is a specific type of tear in the meniscocapsular junction of the posterior horn of the medial meniscus, usually associated with anterior cruciate ligament (ACL) injury. Biomechanical cadaveric studies have shown that ACL injury combined with ramp lesions significantly increases anterior tibial translation and external rotation, which ACL reconstruction alone cannot completely control. Additionally, ramp lesions are sometimes associated with medial meniscal defects, especially in cases of chronic ACL deficiency after repetitive traumatic events, in which the anatomical repair of the meniscocapsular junction is infeasible. This report describes a new arthroscopic repair technique using an all-suture anchor through a posteromedial portal for unstable ramp lesions with medial meniscal defects.

Comparison of Foxp3+ regulatory T cells and CD163+ macrophages in invasive and non-invasive extramammary Paget's disease.

Regulatory T cells (Tregs), identified by the expression of CD4, CD25 and Foxp3, together with immunosuppressive macrophages, such as CD163+ M2 macrophages, are involved in maintaining peripheral tolerance. The aim of this study was to elucidate the involvement of Tregs and CD163+ macrophages in invasive and non-invasive extramammary Paget's disease. The presence of CD4+CD25+Foxp3+ Tregs, CD163+ M2 macrophages and matrix metalloproteinase-9+ cells was examined immunohistologically in fixed sections of lesional skin from 10 patients with non-invasive extramammary Paget's disease and 7 patients with invasive extramammary Paget's disease. Fewer CD4+CD25+Foxp3+ Tregs were observed in non-invasive extramammary Paget's disease than in invasive extramammary Paget's disease. In contrast, higher numbers of CD163+ macrophages and metalloproteinase-9+ cells were detected only in invasive extramammary Paget's disease. These findings suggest that the induction of immunosuppressive cells in extramammary Paget's disease differs according to the tumour stage.

[An autopsy case of extraskeletal osteosarcoma arising in the mediastinum].

A 90-year-old woman was admitted to our hospital with dyspnea and palpitation. A chest X-ray film showed an enlarged cardiac outline. An enhanced chest CT scan showed a 35-mm tumor in the superior portion of the mediastinum, a large amount of pericardial effusion, and bilateral lung nodules. She showed progressive deterioration and died 2 weeks after admission. At autopsy, there was a solid tumor measuring 35 mm in the anterior mediastinum between the aortic arch and trachea. The tumor invaded the left subclavian artery. The pathological autopsy finding was extraskeletal osteosarcoma arising from the mediastinum. Although extraskeletal osteosarcoma is uncommon and rarely develops in the mediastinum, it should be included in the differential diagnosis of a mediastinal mass.

[A case of negative pressure pulmonary hemorrhage associated with upper-airway obstruction due to a giant vocal cord nodule].

A 68-year-old woman was brought by ambulance because of sudden choking sensation and dyspnea after coughing. A physical examination showed hoarseness and blood-stained sputum. A chest computed tomography (CT) showed bilateral diffusely-distributed ground-glass opacity. A bronchoscopy examination revealed a large nodule on the left larynx and bloody bronchoalveolar lavage fluid. We diagnosed negative pressure pulmonary hemorrhage (NPPH) based on suspected upper-airway obstruction, but it resolved without further treatment. We found no other causes. We report a rare case of NPPH due to a giant vocal cord nodule.

Scapular Winging following Sports-Related Injury in a Rugby Player.

The most common cause of medial scapular winging is long thoracic nerve palsy (LTN) and subsequent serratus anterior muscle dysfunction. A 16-year-old right-handed male high-school rugby player developed severe right-sided neck and shoulder pain after tackling an opponent while playing rugby. Six weeks after initial injury, the patient observed shoulder muscle weakness when performing his daily activities. On physical examination, limited active elevation of the right shoulder in the scapular plane and scapular winging was observed. Magnetic resonance imaging revealed atrophy of both the SA and subclavius muscles on the right side, and we initially suspected an LTN injury sustained. However, while detailing his history, the patient explained that he also had noted difficulty sucking high viscosity drinks such as shakes and smoothies since childhood. In addition, physical examination showed weakness of the orbicularis oculi muscle. Considering the facial muscle weakness, facioscapulohumeral dystrophy (FSHD) was also suspected, and genetic testing showed chromosome 4q35 deletion with restriction fragments 17 kb and 3 tandem repeated DNA confirming the diagnosis of FSHD. Clinicians should be aware that FSHD could be one of the differential diagnoses of scapular winging after sports injury, and surgeons should rule out the diagnosis of FSHD before performing any surgical treatment for SA palsy.

4-Methylumbelliferone suppresses catabolic activation in anterior cruciate ligament-derived cells via a mechanism independent of hyaluronan inhibition.

BACKGROUND: The anterior cruciate ligament (ACL) has a key role as a dynamic stabilizer of the knee joints, and ACL dysfunction caused by traumatic or degenerative rupture accelerates osteoarthritis progression. Thus, it is important to prevent the degenerative rupture of the ACL. 4-Methylumbelliferone (4-MU), a pre-approved drug, exerts anti-inflammatory effects in osteoarthritis chondrocytes. It was originally used as an inhibitor of hyaluronan synthesis in chondrocytes. METHODS: In this study, we investigated whether 4-MU affects the expression of catabolic factors, such as matrix metalloproteinase (MMP)-1, MMP-3, and interleukin (IL)-6, in ACL-derived cells and ACL explant cultures using immunohistochemistry, real-time RT-qPCR, and capillary western immunoassay. Furthermore, the hyaluronan concentration was evaluated using a colorimetric assay. Statistical analyses were conducted using analysis of variance for multi-group comparisons, followed by Tukey or Tukey-Kramer post hoc test. RESULTS: Our results revealed, for the first time, that 4-MU suppressed the IL-ß-induced upregulation of pro-catabolic factors, such as MMP-1, MMP-3, and IL-6, in ACL-derived cells. This suppressive effect was also observed in the cultured ligament tissues in ex vivo experiments. 4-MU also reversed an enhanced dependence on glycolysis in IL-1ß-activated ACL-derived cells. Furthermore, we found that the suppressive effects of 4-MU were exerted directly and not through the inhibition of hyaluronan synthesis. CONCLUSIONS: We conclude that 4-MU could be an effective and useful treatment for knee osteoarthritis, owing to its anti-inflammatory effect on, not only chondrocytes but also on ligament cells.

[A rare case of large cell neuroendocrine carcinoma of the thymus].

A 46-year-old man presented with chest pain at a local hospital in July 2007. Chest computed tomography (CT) showed a 48-mm mass in the anterior mediastinum. CT-guided percutaneous tumor biopsy demonstrated large cell neuroendocrine carcinoma of the thymus. He was referred to our hospital in August 2007. Because the tumor had already progressed to stage IVb according to the Masaoka classification of thymic epithelial tumors, the patient was treated with combination chemotherapy of cisplatin and irinotecan, which achieved a partial response. However, the tumor relapsed in February 2008. He died, despite 2 separate cycles of chemotherapy with docetaxel only and amrubicin only in August 2008. We encountered a rare case of large cell neuroendocrine carcinoma of the thymus treated with combination chemotherapy of cisplatin and irinotecan.

[Case of benign asbestos pleurisy with diffuse pleural thickening confirmed on autopsy].

We report a 65-year-old man with a 35-year history of occupational asbestos exposure. He presented at a nearby hospital with a complaint of dyspnea in 2002. Bilateral pleural effusion was revealed on a chest x-ray film. Chest CT revealed diffuse thickening of the pleura, bilateral pleural effusions and cardiac effusion, but no abnormal findings in the lung fields. Both pleural effusions were exudative, and lymphocytes were predominant. Antituberculous chemotherapy had no effect on the exudates. Thoracoscopic pleural biopsy was conducted to exclude malignant mesothelioma. No evidence of malignancy was found in pleural samples. The patient's condition was diagnosed as benign asbestos pleurisy with diffuse pleural thickening. He was referred to our hospital in June 2008. Bilateral pleural effusions continued to progress despite pleurodesis and frequent drainage of his pleural effusion. He suffered from respiratory failure and died in December 2008. We investigated the concentration of asbestos bodies in his lung tissue. There were 462 asbestos bodies per 1 g of dry lung tissue, which was relatively low considering the time of asbestos exposure. We report a rare case of benign asbestos pleurisy with diffuse pleural thickening confirmed by autopsy.