Anticoagulant Use as an Independent Risk Factor and Higher In-Hospital Mortality in Patients Showing Alveolar Hemorrhage in Diffuse Lung Disease.

Background and objectives: Bronchoalveolar lavage (BAL) is commonly performed to evaluate diffuse lung disease and occasionally to identify alveolar hemorrhage. However, the clinical impact of alveolar hemorrhage and its risk factors in patients with diffuse lung disease have not been clarified. Materials and Methods: We retrospectively analyzed the medical records of all patients who underwent BAL to evaluate diffuse lung disease from January 2017 to December 2020. Alveolar hemorrhage was defined as progressive hemorrhagic BAL fluid or the presence of ≥20% hemosiderin-laden macrophages in the BAL fluid. Logistic regression analysis was performed to assess the association between alveolar hemorrhage and other factors. Results: Sixty subjects were enrolled in this study. Alveolar hemorrhage was observed in 19 subjects (31.7%) with idiopathic interstitial pneumonia, acute respiratory distress syndrome, interstitial pneumonia with autoimmune features, drug-induced lung injury, eosinophilic pneumonia, adenocarcinoma, and systemic lupus erythematosus. The use of anticoagulants was a significant risk factor for alveolar hemorrhage (odds ratio 7.57, p = 0.049). Patients with alveolar hemorrhage required intubated mechanical ventilation more frequently (63.2% vs. 24.4%, p = 0.005) and had higher in-hospital mortality rates (26.3% vs. 4.9%, p = 0.028) than those without alveolar hemorrhage. Conclusions: Alveolar hemorrhage was observed in various etiologies. The use of anticoagulants was a significant risk factor for alveolar hemorrhage. Patients with alveolar hemorrhage showed more severe respiratory failure and had higher in-hospital mortality than those without alveolar hemorrhage.

[Cystic fibrosis with focal biliary cirrhosis and portal hypertension in Japan: a case report].

A 9-year-old Japanese girl was found to have persistently elevated hepatic enzymes, chronic bronchitis, chronic sinusitis, and poor weight gain beginning at 5 months of age. Chest computed tomography (CT) revealed diffuse bronchial wall thickening and peripheral bronchiectasis. Abdominal CT showed pancreatic atrophy, liver cirrhosis, a dilated splenic vein, and splenomegaly. Her sweat chloride concentration was 117mmol/l (normal, <60mmol/l). CFTR gene analysis revealed the presence of the Y517H variant on one allele and the 1540del10 variant one the other allele. These findings established a definitive diagnosis of cystic fibrosis (CF). While CF is the most common autosomal recessive genetic disorder among Europeans, it is quite rare in Southeast Asia including Japan. It is important that CF be considered in the work-up of children with chronic hepatic and respiratory disorders even if it is uncommon among children of a similar background.

Potential complications in patients undergoing an ethanol injection into the vein of Marshall.

BACKGROUND: Ethanol injections into the vein of Marshall (VOM) (EIM) are considered to be a good therapeutic option for atrial tachyarrhythmias, however, the safety remains to be determined. To elucidate what would affect the safety and potential complications of an EIM, we investigated the anatomical features of the VOM and patient background. METHODS: We performed the EIM before the conventional pulmonary vein isolation for drug-resistant atrial fibrillation in 88 patients and evaluated the anatomical features of the VOM and their background. RESULTS: All procedures were completed, however, other than myocardial staining, trivial contrast medium leaked out of the VOM into the pericardial space, that is, extravasation of contrast medium with capillary rupture, during the EIM in 20 patients (22.7%) regardless of the features of the VOM. No pericardial effusions requiring further intervention developed after the extravasation, which resolved by the next day on echocardiography in 18 of those patients. However, two patients who had extravasation other than during the initial contrast injection required additional therapeutic intervention for nonnegligible pericardial effusions. Their body weights were significantly lower and the latter two patients were also small lean women with heart failure and a preserved ejection fraction. CONCLUSIONS: The physical constitution, regardless of the characteristics of the VOM, could be strongly associated with adverse events during the EIM. We must take extreme care in smaller patients with poor compliant hearts during the EIM.

[Pulmonary Carcinosarcoma Presenting Hemothorax Caused by Pleural Invasion;Report of a Case].

A 71-year-old man presented with hemothorax with cough, sputa and worsening dyspnea. On chest X-ray and computed tomography(CT), a huge tumor in the right upper lobe with hematoma and small amount of gas suggesting hemopneumothorax was revealed. No apparent lymphadenopathy nor intrapulmonary metastases were observed. The tumor showed a little enhancement on the contrastenhanced CT. Then the resction of the tumor was performed, and the pathological evaluation revealed a carcionosarcoma (adenocarcinoma+osteosarcoma) pT3N0 (stage II B) G4 pl2. Sarcomatoid carcinoma such as carcinosarcoma should be considered as a possible cause of hemothorax in making a diagnosis of hemorrhagic hypovascular huge lung tumor.

A Case of Myocardial Infarction with Non-obstructive Coronary Arteries Complicated by a Ventricular Septal Rupture: The Challenge of Achieving Hemodynamic Stability Using Impella Devices.

The prognosis of myocardial infarction with non-obstructive coronary arteries (MINOCA) is reported to be as worse as myocardial infarction with obstructive coronary arteries. However, its mechanical complications are still poorly understood. A 71-year-old woman developed MINOCA after ascending aortic replacement surgery. During treatment, the patient experienced cardiogenic shock due to a ventricular septal rupture (VSR). The introduction of Impella devices reduced the left-to-right shunt and improved the patient's hemodynamics. Finally, a scheduled surgical repair was performed under stable conditions. In this report, we focused on the pathophysiology of MINOCA-related VSR and discussed the effectiveness of Impella devices as a bridge to surgical repair and circulatory backup during the perioperative period.

Clinicopathological significance of MAML2 gene split in mucoepidermoid carcinoma.

CRTC1-MAML2 and CRTC3-MAML2 fusions have been associated with favorable clinicopathological features of mucoepidermoid carcinomas. However, the significance of the MAML2 gene split has not been fully clarified. In the present study, 95 mucoepidermoid carcinomas (paraffin-embedded materials) were analyzed for CRTC1-MAML2 and CRTC3-MAML2 fusions by RT-PCR and for the MAML2 gene split by FISH. Quantitative RT-PCR for the CRTC1-MAML2 transcript was performed in selected cases. MLL gene involvement, which has been reported in some leukemia cases, was examined by FISH in fusion partner-unknown cases. CRTC1-MAML2 and CRTC3-MAML2 fusions were detected in 37 and 6 cases, respectively. The MAML2 gene split was detected in 62 cases, which included all CRTC1/3-MAML2 fusion-positive cases. The level of CRTC1-MAML2 transcript expression was highly variable, and its clinicopathological impact was unclear. The MLL gene split was not detected. Mucoepidermoid carcinomas negative for CRTC1/3-MAML2 and positive for the MAML2 gene split (n = 19) showed favorable clinicopathological tumor features similar to those positive for CRTC1/3-MAML2 fusions. Compared with negative cases (n = 33), mucoepidermoid carcinomas positive for the MAML2 split (n = 62) were associated with lower patient age, a mild female predilection, a smaller tumor size, less frequent nodal metastasis, a lower clinical stage, a lower histological grade, and longer overall and disease-free survival. The MAML2 gene split emerged as an independent prognostic factor for both overall and disease-free survival in multivariate prognostic analysis. The presence of the MAML2 gene split defines a distinct mucoepidermoid carcinoma subset that is associated clinicopathologically with favorable tumor features.

Characterization of chromosomal aberrations in thymic MALT lymphoma.

Mucosa-associated lymphoid tissue (MALT) lymphoma arising in the thymus is a rare disorder that shows a strong association with autoimmune disease. Several MALT-lymphoma-specific and -associated chromosomal abnormalities, including t(11;18), t(14;18), t(1;14), trisomy 3 and trisomy 18, are known to occur. The former translocation results in apoptosis inhibitor 2 gene (API2)-MALT lymphoma-associated translocation 1 (MALT1) fusion. In this study, we examined 14 cases of thymic MALT lymphomas for API2-MALT1 fusion using multiplex reverse transcription polymerase chain reaction and looked for trisomy 3, trisomy 18 and abnormalities of MALT1 and IGH genes using fluorescence in situ hybridization. Thymic MALT lymphoma cases had a high frequency of trisomy 3 (7/14 cases), a very low incidence of trisomy 18 (1/14) and no detectable MALT1-associated (0/13) or IGH-associated (0/13) gene abnormalities including t(11;18). A review of the literature showed that the pattern of chromosomal aberrations in thymic MALT lymphoma was similar to those of thyroid and salivary gland MALT lymphomas. Although frequently detected, trisomy 3 was not associated with any of the clinicopathological factors analyzed, suggesting that trisomy 3 may play a role in lymphoma development. In conclusion, the present study showed that thymic MALT lymphoma has a characteristic pattern of chromosomal aberrations that may be similar to those of other autoimmune-associated MALT lymphomas.

Total synthesis and antimicrobial evaluation of (+)-hygrophorone B12 and its analogues.

This paper describes the synthesis and evaluation of lead compounds with a new chemical skeleton that is not found in conventional antimicrobial agents. The biologically attractive cyclopentenoid (+)-hygrophorone B12, isolated from the fruiting bodies of Hygrophorus abieticola, and its analogues were synthesized in a longer linear sequence of twelve steps, starting from a cyclopentenone derivative. This synthesis involved the following crucial steps: (i) oximation of a ketone to stabilize the requisite aldehyde to install a side chain and (ii) coupling of an aldehyde with a side chain to assemble the desired hygrophorone. Then, the antimicrobial activity of these hygrophorones towards clinically relevant bacterial pathogens was evaluated. The results showed that hygrophorone B12 and its analogues are especially effective in preventing the proliferation of gram-positive bacteria. In addition, it was found that some structural features such as the presence of the enone moiety as well as the carbon-carbon triple bond on the hydrocarbon chain were pivotal to increase the antimicrobial activity of hygrophorone B. This study is expected to support the development of novel antimicrobial agents by flexibly synthesizing hygrophorone B analogues with a carbon five-membered ring skeleton from the common intermediate.

Erratum to "Ependymoma of the broad ligament mimicking an ovarian surface epithelial tumor" [Radiology Case Reports 16 (2021) 210-214].

[This corrects the article DOI: 10.1016/j.radcr.2020.10.046.].

Hemoptysis after coil embolization for pulmonary arteriovenous malformation: Histopathological confirmation of bronchial epithelium extension.

Coil embolization is widely performed for pulmonary arteriovenous malformations (PAVMs). We describe herein 2 cases of hemoptysis during long-term follow-up after coil embolization for PAVMs. For both cases, lobectomy was performed and histopathological examinations revealed chronic inflammation and bronchial epithelium extension into the sac of the PAVM. In addition, we performed a systematic review of previous reports of hemoptysis after embolization for PAVMs.

Impact of CRTC1/3-MAML2 fusions on histological classification and prognosis of mucoepidermoid carcinoma.

AIMS: The aim of study was to evaluate the impact of CRTC1-MAML2 and CRTC3-MAML2 fusions on the histological classification of mucoepidermoid carcinoma (MEC) of the salivary glands and on the prognosis of patients. METHODS AND RESULTS: MEC cases (n = 111) were screened for CRTC1-MAML2 and CRTC3-MAML2 fusions by reverse transcription polymerase chain reaction. We developed a system of 'molecular Armed Forces Institute of Pathology (AFIP) classification' that combined the AFIP histological classification proposed by Goode et al. and the presence of CRTC1-MAML2 or CRTC3-MAML2 fusions. MEC cases positive for CRTC1-MAML2 or CRTC3-MAML2 fusion formed a favourable tumour subset that was distinct from fusion-negative cases. When positive for the fusions, 'high-risk' patients, including those with a higher histological grade or an advanced clinical stage, showed an excellent prognosis. For overall survival, 'molecular AFIP classification' was selected as a powerful independent prognostic factor (P=0.0038), as was the clinical stage (P =0.0032). For disease-free survival, 'molecular AFIP classification' was also selected as an independent prognostic factor (P = 0.0006). CONCLUSIONS: Molecular AFIP classification may be useful in predicting the prognosis of patients with MEC.

Myelolipoma mimicking osteosarcoma in the distal femur.

INTRODUCTION AND IMPORTANCE: Periosteal reactions indicate malignant bone tumors, including osteosarcoma; establishing an accurate diagnosis is key to determining the most appropriate treatment strategy. We describe a rare case of myelolipoma in the distal femur metaphysis with massive extraskeletal lesions and periosteal reactions. CASE PRESENTATION: A 25-year-old woman was referred to our hospital to treat a gradually expanding mass around her knee that grew to the size of a baby's head. She had a history of hydrocephalus caused by congenital cytomegalovirus infection and was bedridden for life. Radiography showed a prominent osteoblastic rim and osteolytic lesion with a moth-eaten appearance. Osteosarcoma was suspected due to excessive extraskeletal invasion and periosteal reactions. T1- and T2-weighted magnetic resonance images showed a high-signal-intensity homologous lesion. Biopsy specimens contained adipose and hematopoietic tissues. A myelolipoma was diagnosed. Due to her fragility, surgical intervention was suspended. Two years after diagnosis, the tumor size did not change. CLINICAL DISCUSSION: Myelolipomas are benign tumors that typically arise from the adrenal gland and rarely develop in the extremities. This type of tumor typically does not cause any tumor-related symptoms or endocrine disturbances and has been reported as a type of incidentaloma. To effectively manage myelolipoma patients, differential diagnosis of tumors mimicking malignant bone tumors is important. CONCLUSION: We successfully managed a destructive ectopic myelolipoma in the distal femoral metaphysis, with massive extraskeletal lesions and periosteal reactions. Clinicians should appropriately differentiate myelolipoma from tumors mimicking malignant bone tumors.

Examination on the necessity of pericardial fat tissue resection in extended thymectomy for myasthenia gravis.

BACKGROUND: Extended thymectomy with pericardial fat tissue resection has been a mainstay in the treatment for myasthenia gravies (MG), but few studies have examined the necessity of the pericardial fat tissue resection in extended thymectomy. METHODS: We pathologically examined the distribution of germinal centers in the resected thymus including the thymus-surrounding fat tissue. Patients who underwent extended thymectomy using subxiphoid thoracoscopy for generalized MG or thymoma with anti-acetylcholine receptor antibody (anti-AchR Ab) positivity from March 2015 to January 2021 were included in this study. RESULTS: A total of 20 patients underwent extended thymectomy (generalized nonthymomatous MG, n=5; generalized MG with thymoma, n=6; thymoma with anti-AchR Ab positivity, n=9). The resected specimens were divided to 6 parts and were pathologically examined to investigate the distribution of the geminal centers in all lesions. The number and distribution of germinal centers in the thymus varied depending on the patient, and no germinal centers were identified in the right or left pericardial fat tissues. CONCLUSIONS: It is necessary to resect the pericardial fat tissue in patients whose preoperative images show the presence of pericardial fat tissue hyperplasia or ectopic thymoma. However, extensive pericardial fat tissue resection might not be necessary for all MG patients who undergo extended thymectomy with opening of the bilateral pleural by video-assisted or robot-assisted thoracoscopic surgery (RATS) via a subxiphoid approach.

Type A thymoma with simultaneous solitary intrapulmonary metastasis: A case report.

A 79-year-old woman was referred to our facility because of an abnormal chest shadow. Chest computed tomography (CT) showed a solitary right middle lung nodule with a maximum diameter of 3 mm and anterior mediastinal nodule with a maximum diameter of 21 mm. The lung nodule was suspected of being a primary lung cancer rather than a metastatic tumor because there were no primary malignant tumors, apart from an anterior mediastinal tumor visible on diagnostic imaging, including F18 fluorodeoxyglucose-positron emission tomography, and a solitary lung nodule. Partial lung resection by video-assisted thoracoscopic surgery (VATS) was performed, and the intraoperative frozen section of the tumor tissue resulted in a diagnosis of carcinoid tumor. As a result, right middle lobectomy by VATS was performed. The final histological diagnosis of the permanent specimen was intrapulmonary type A thymoma. VATS thymectomy was performed three months later. The histological diagnosis was type A thymoma with intrapulmonary metastasis (Masaoka stage IVb). Additional therapy was not performed because complete resection was achieved. Follow-up CT was performed once every six months after the operation. The patient has been followed up for one year without any further recurrence.

Ependymoma of the broad ligament mimicking an ovarian surface epithelial tumor.

We report a case of ependymoma of the broad ligament occurring in a 21-year-old woman. CT and MRI findings showed a 40-mm-diameter, well-demarcated cystic mass with a lobulated solid component in the right pelvis. The solid component showed heterogeneous intermediate signal intensity on T2-weighted image and prolonged mild contrast enhancement. The tumor was resected and confirmed as ependymoma based on the histologic findings along with its immunohistochemical profile. To our knowledge, this is the first report of an adnexal ependymoma describing the precise radiological characteristics that resembled those of borderline or malignant epithelial ovarian tumors.

A Case of Alveolar Haemorrhage Induced by Iodinated Contrast Medium Use.

An 81-year-old man complaining of exertional dyspnoea underwent coronary angiography using an iodinated contrast medium. After angiography, the patient required systemic corticosteroid therapy because of respiratory failure due to alveolar haemorrhage. Percutaneous coronary intervention was performed 29 days after angiography using the same contrast medium. After the intervention, the patient required intubated mechanical ventilation and renal replacement therapy. Bronchoalveolar lavage was bloody with many haemosiderin-filled macrophages. Systemic corticosteroid therapy again improved his clinical condition. Iodinated contrast media may cause alveolar haemorrhage and re-exposure to contrast media may induce a more severe adverse reaction. LEARNING POINTS: Iodinated contrast media may cause alveolar haemorrhage.Re-exposure to iodinated contrast media may induce a more severe adverse reaction.

The inflammatory pseudotumor presenting periodic acid-Schiff-positive inclusions with acute unilateral facial nerve palsy.

Although most acute peripheral facial palsies can be attributed to Bell's palsy, other factors, such as infection, trauma, and neoplasm, can cause facial palsy as well. Among these, facial nerve tumors are rare but should be considered in the differential diagnosis of facial palsy. Palsies due to facial nerve tumors usually present with slow onset but occasionally present as acute episodes. In such cases, facial nerve decompression is the treatment of choice to allow the tumor room to grow without compressing the nerve or its blood supply. We describe a case of severe, acute facial palsy presenting with a spindle-shaped bone erosion on the mastoid portion of the facial canal. Although facial neuroma was suspected preoperatively, emergency decompression surgery revealed that an unusual inflammatory pseudotumor was responsible for the finding. Postoperative histological analysis revealed extensive destruction of the nerve fibers, with extensive infiltration of foamy macrophages containing characteristic, diastase-resistant, periodic acid-Schiff (PAS)-positive inclusions, which are hallmark of the uncommon bacterial infections. This was a case of facial palsy with an unusual etiology. The case shows the benefit of decompression surgery not only as treatment for the palsy but also as exploratory surgery in cases of facial nerve tumor.

Testicular teratoma demanded in-depth pathological exploration to rule out malignancy: A pediatric case report.

INTRODUCTION: Prepubertal testicular tumors can be both benign and malignant. Although most testicular teratomas are benign, some immature cases include malignant transformation or the mixed type with yolk sac tumor and, occasionally, it is challenging to rule out malignancy. CASE PRESENTATION: We present a case of immature testicular teratoma in a 7-month-old infant, whose alpha-fetoprotein level was sequentially elevated following orchidectomy. Since malignancy could not be ruled out, we performed whole body imaging and in-depth pathological exploration. GLYPICAN3, OCT3/4, and SOX2 staining revealed no evidence of malignancy. The patient was finally diagnosed with benign immature teratoma, and has been free from recurrence for 3 years. CONCLUSION: Here, we describe the case report, as well as all the comprehensive diagnostic tests that we performed in order to rule out the malignant component.

Sinonasal inverted papilloma associated with small cell neuroendocrine carcinoma: A case report and literature review of rare malignancies associated with inverted papilloma.

We report a rare case of sinonasal inverted papilloma (IP) associated with small cell neuroendocrine carcinoma (SNEC). To our knowledge, this is the first report to describe SNEC found during the treatment of sinonasal IP. Surgery and five cycles of cisplatin plus etoposide with concurrent intensity modulated radiation therapy were performed. Neither local recurrence nor distant metastasis was noted during 6 years of post-diagnostic follow-up. The prognosis of SNEC is very poor. Treatment planning for sinonasal IP should consider a possible association with this rare but aggressive malignancy, whose treatment is completely different from that of squamous cell carcinoma, a malignancy which is commonly associated with IP. We also performed a PubMed review of the literature to identify the incidence and pathological diagnosis of associated malignancy. Among a total of 5286 cases of sinonasal IP (61 studies), the incidence of associated malignancy was 8.02% in squamous cell carcinoma, 0.19% in transitional cell carcinoma, 0.04% in mucoepidermoid carcinoma, 0.02% in verrucous cell carcinoma and 0.02% in adenocarcinoma. The incidence of associated malignancy was significantly higher in East and Southeast Asia (11.0%) and North America (10.4%) than in Europe (3.9%) (p=0.04 and p=0.03, respectively; T-test).

Simultaneous determination of intraparticle diffusivities from ternary component uptake curves using the shallow bed technique.

In order to design and operate a fixed-bed reactor, accurate modeling is important. For a single component system, the determination of intraparticle diffusivity is rather easy. However, the calculations of multi-component systems are normally complicated and very time-consuming. Therefore, an alternative simple determination procedure using the shallow bed technique is proposed in this research to determine the intraparticle diffusivities for multi-component systems. Ternary component systems of phenol (PH), benzoic acid (BA), and p-nitro-phenol (PNP) were investigated as model components. This study illustrated that adsorption uptake curves of different components in ternary systems can be converted into one typical characteristic curve (theoretical uptake curve, TUC) by using a set of dimensionless groups. By matching the dimensionless experimental uptake curve (DEUC) with TUC, diffusivities of PH, BA and PNP were determined as 8.00 x 10(-8), 5.92 x 10(-8) and 5.05 x 10(-8)cm2 s(-1), respectively. These values are in good agreement with simulated experimental values. This study demonstrated that the shallow bed technique can be used to simultaneous determination of intraparticle diffusivities from multi-component systems.