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Duchenne muscular dystrophy (DMD) is the most common type of muscular dystrophy, but the spinal cord is rarely examined. Here we report a case of DMD with interesting spinal cord findings. In a 37-year-old man with DMD accompanied by hypoxic encephalopathy from the age of 32 years, autopsy showed amyotrophic lateral sclerosis-like pyramidal tract degeneration over the entire spinal cord, presumably due to hypoxic encephalopathy. Furthermore, anterior horn cells exhibited Wallerian degeneration-like changes. To investigate more about the pathogenesis, an immunohistochemical study using anti-synaptophysin, glutamic acid decarboxylase (GAD), postsynaptic density protein-95 (PSD-95) and choline acetyltransferase (ChAT) antibodies was performed. Immunostaining for synaptophysin showed that the number of synapses around anterior horn cell were decreased, contrary to the finding of teenage DMD, in which the number of synapses were increased, probably due to the reaction toward the reduced anterior horn cell activity. The decrease of synapses of the present case may be mainly due to hypoxic encephalopathy, based on the degeneration of the pyramidal tract. Another interesting finding is that GAD was strongly positive in the cytoplasm of anterior horn cells, which may be explained by Wallerian degeneration-like mechanism. Moreover, the expression of PSD-95 is increased in anterior horn cells. Compensation for postsynaptic damage can be considered, since dystrophin is necessary for maintaining the post-synaptic maintenance. There were no apparent differences in ChAT immunostaining. Further investigation is necessary whether these findings are characteristic of long-term surviving cases accompanied by hypoxic encephalopathy.

Abstract One of the resistant mechanisms of EGFR‐TKIs is BRAF V600E mutation. Herein, we present the case of a 54‐year‐old Japanese female who underwent a right middle lobectomy for pathological stage IIB lung adenocarcinoma. One year and nine months after the surgery, she developed multiple intrapulmonary metastases. Osimertinib was administered due to EGFR exon 19 deletion. Although all intrapulmonary metastases had shrunk, the nodule at the superior segment of left lung enlarged after postoperative 4 years. The tumour was resected and BRAF V600E mutation and exon 19 deletion were detected. Three months after treatment with dabrafenib and trametinib instead of osimertinib, the remaining intrapulmonary metastases increased again. The continued growth of the metastatic foci even after EGFR‐TKI may indicate an acquired resistance. Thus, a repeat biopsy will aid in confirming the new gene expression. It should have been necessary to administer an additional dose of dabrafenib and trametinib without discontinuing osimertinib.

Adipose tissue-derived non-esterified saturated long-chain fatty acid palmitate (PA) decisively contributes to β-cell demise in type 2 diabetes mellitus in part through the excessive generation of hydrogen peroxide (H2O2). The endoplasmic reticulum (ER) as the primary site of oxidative protein folding could represent a significant source of H2O2. Both ER-oxidoreductin-1 (ERO-1) isoenzymes, ERO-1α and ERO-1β, catalyse oxidative protein folding within the ER, generating equimolar amounts of H2O2 for every disulphide bond formed. However, whether ERO-1-derived H2O2 constitutes a potential source of cytotoxic luminal H2O2 under lipotoxic conditions is still unknown. Here, we demonstrate that both ERO-1 isoforms are expressed in pancreatic β-cells, but interestingly, PA only significantly induces ERO-1α. Its specific deletion significantly attenuates PA-mediated oxidative ER stress and subsequent β-cell death by decreasing PA-mediated ER-luminal and mitochondrial H2O2 accumulation, by counteracting the dysregulation of ER Ca2+ homeostasis, and by mitigating the reduction of mitochondrial membrane potential and lowered ATP content. Moreover, ablation of ERO-1α alleviated PA-induced hyperoxidation of the ER redox milieu. Importantly, ablation of ERO-1α did not affect the insulin secretory capacity, the unfolded protein response, or ER redox homeostasis under steady-state conditions. The involvement of ERO-1α-derived H2O2 in PA-mediated β-cell lipotoxicity was corroborated by the overexpression of a redox-active ERO-1α underscoring the proapoptotic activity of ERO-1α in pancreatic β-cells. Overall, our findings highlight the critical role of ERO-1α-derived H2O2 in lipotoxic ER stress and β-cell failure.

Abstract Mucosa‐associated lymphoid tissue (MALT) is a low‐grade lymphoma, but cases in which it has transformed into a high‐grade lymphoma have been reported, necessitating an accurate diagnosis. The patient was a 79‐year‐old nonsmoking Japanese female with history of ocular sarcoidosis. A computed tomography scan of her chest revealed a 35‐mm nodule in the left S1 + 2, contiguous with the lymph nodes. Additional nodules were observed around the left B5 and B10a. Bronchoscopy revealed stenosis caused by a white, glossy, elevated lesion with angiogenesis at the orifice of the left upper lobe bronchus. The biopsy specimen demonstrated the dominance of lymphoid cells and tested positive for CD20, CD79a, Bcl‐2, and IRTA‐1, which is consistent with the findings in MALT lymphoma. Therefore, in the presence of multiple infiltrative shadows along the bronchi with glossy elevated lesions without necrosis on bronchoscopy, it is important to consider MALT lymphoma as a differential diagnosis.

Abstract Background Neurofibromatosis type 1 (NF1) is known to be associated with the frequent occurrence of unique gastrointestinal stromal tumors (GISTs), preferably occurring in the small intestine, with no mutations in the c-kit proto-oncogene or platelet-derived growth factor receptor-alpha (PDGFRA), with a high tendency for multifocal development, indolent nature, with low proliferation activity and favorable prognosis. Case presentation A woman in her forties visited her local doctor complaining of menstrual pain; a large mass was detected in her lower abdomen, and she was referred to our hospital. The patient had hundreds of skin warts and café au lait spots. The patient’s mother had been diagnosed with type 1 neurofibromatosis. The patient met the diagnostic criteria for NF1 and was diagnosed with NF1. Ultrasonography showed a large heterogeneous cystic mass with various echo patterns, solid compartments and multiple septations. Magnetic resonance imaging showed a multilocular cystic mass with liquid content exhibiting various intensities, including that of blood. A small round solid mass was also observed close to the cystic tumor. Contrast-enhanced computed tomography showed that the round solid mass showed strong enhancement in the early phase, unlike the cystic tumor component. Open laparotomy revealed a multicystic exophytic tumor measuring 11.5 cm originating from the jejunal wall, 20 cm distal to the duodenojejunal flexure. A solid tumor measuring 2.1 cm was also found on the anal side of the large tumor. We resected the short segment of the jejunum, including the two lesions. Microscopic findings revealed that the cystic and solid tumors consisted of spindle-shaped tumor cells showing little atypia with a fascicular or bundle arrangement. Nuclear mitosis was scarce. Immunostaining of the tumor cells showed positive staining for KIT and DOG1 and negative staining for S100 and desmin. The NF1 patient was diagnosed with multiple GISTs accompanied by intratumoral hemorrhagic denaturation arising from the jejunum. The TNM staging was pT4N0M0, stage IIIA. Conclusion We report a case of GISTs associated with NF1 that showed a jejunal origin, multifocal development and few mitotic figures. The recurrence risk, survival prognosis and need for adjuvant chemotherapy, particularly in cases where the initial GIST exhibits a very indolent pathology in NF1-related GISTs, remain to be elucidated.

Abstract Background Alteration of chemosensitivity or tumor aggressiveness in response to chemotherapy has been reported, and liquid biopsy assessment during chemotherapy for colorectal cancers has confirmed the acquisition of mutations in various oncogenes. However, the occurrence of histological transformation seems to be extremely rare in colorectal cancers, and the few existing case reports of this transformation are from lung cancer and breast cancer. In this report, we describe the histological transformation of clinically aggressive scirrhous-type poorly differentiated adenocarcinoma of the ascending colon to signet-ring cell carcinoma in almost all recurrent tumors that were confirmed by autopsy after response to chemotherapy plus cetuximab. Case presentation A 59-year-old woman visited our hospital with whole abdominal pain and body weight loss and was diagnosed with scirrhous-type poorly differentiated adenocarcinoma of the ascending colon with aggressive lymph node metastases. The intrinsic chemosensitivity of the tumors was evident upon initiation of mFOLFOX6 plus cetuximab therapy, and right hemicolectomy was performed, and the tumor obviously remained in the peripancreatic area, paraaortic region, or other retroperitoneal areas. The ascending colon tumors mainly consisted of poorly differentiated adenocarcinoma and were not associated with signet-ring cell components except for minute clusters in a few lymphatic emboli in the main tumor. Chemotherapy was continued, and metastases were eliminated at 8 months after the operation; this response was maintained for an additional 4 months. Discontinuation of chemotherapy plus cetuximab resulted in immediate tumor recurrence and rapid expansion, and the patient died of the recurrent tumor 1 year and 2 months after the operation. Autopsy specimens revealed that almost all of the recurrent tumors exhibited transformation and consisted of signet-ring cell histology. Conclusion This case might suggest that various oncogene mutations or epigenetic changes resulting from chemotherapy, especially regimens that include cetuximab, contribute to the transformation of non-signet-ring cell colorectal carcinoma to signet-ring cell carcinoma histology and can promote the aggressive clinical progression characteristic of signet-ring cell carcinoma.

We herein report an autopsy case of a fulminant Clostridium perfringens (C. perfringens or Welch bacilli) infection in a healthy adult. A 72-year-old, immunocompetent man visited the emergency department with lower back pain, and blood test revealed hemolytic attack. His condition rapidly worsened with severe acidosis and anemia, and he died despite symptomatic treatment. An autopsy examination demonstrated an abscess with necrosis and air spaces in the right lobe of his liver. Numerous Gram-positive bacilli were seen in the liver and bone marrow, and C. perfringens was identified in culture of the antemortem blood sample. Of note, a mucosal epithelium of the ileum showed loss of tight junctions (claudin 4), suggesting the involvement of C. perfringens toxins with its systemic spreading. Welch toxins were suggested to be involved in serious pathological conditions such as hemolytic anemia and systemic infections, and it is necessary to raise Welch infection as one of the differential diagnoses for fulminant systemic infections even in healthy individuals.

Abstract A 69‐year‐old female Japanese patient presented with an abnormal shadow on chest computed tomography (CT). She had received a mastectomy 14 years prior. Under the diagnosis of primary lung cancer, left upper lobectomy was conducted. Pathology showed a lepidic adenocarcinoma with mediastinal lymph node metastases with pT2aN2M0. Upon retrospective analysis, the chest CT at the time of mastectomy depicted a ground‐glass nodule (GGN) of less than 20 mm. Over the previous 10.5 years, the concentration of the central part of the GGN increased. Conclusively, a pure GGN developed into lung adenocarcinoma with mediastinal lymph node involvement over 14 years. She had bone metastases 4 years after the lobectomy but has survived for five and a half years after surgery with treatment with osimertinib. Comparison readings of films should be performed throughout the patient's clinical history to detect subtle shadow alterations indicative of tumour progression.

Abstract Background Multiple endocrine neoplasia (MEN) is divided into MEN type 1 (MEN-1) and MEN type 2 (MEN-2). MEN-1 may be associated with thymic carcinoid tumors. We present a case of the surgical removal of a thymic carcinoid associated with MEN-1 via a single-incision port RATS. Case presentation A 39-year-old male patient with multiple endocrine neoplasia type 1 (MEN-1) who had an anterior mediastinal mass was referred to our hospital. The patient had undergone total parathyroidectomy and auto-transplantation of a partial parathyroid for hyperparathyroidism 6 years ago. Chest computed tomography revealed an isolated anterior mediastinal mass on the thymic gland with a maximum diameter measuring 22 mm. Thymic carcinoid tumor is classified as MEN-1 and has a poor prognosis, so we decided to remove the tumor. Single-incision port RATS was performed, and thymic carcinoid was confirmed in pathology. Conclusions This report demonstrates that thymic carcinoid tumor removal is feasible and easy to perform via single-incision port RATS.

Abstract Background Pedunculated polyps are more likely to be amenable to complete resection than non-pedunculated early colorectal cancers and rarely require additional surgery. We encountered a patient with a pedunculated early colorectal cancer that consisted of poorly differentiated adenocarcinoma with lymphatic invasion. We performed an additional bowel resection and found nodal metastasis. Case presentation A 43-year-old woman underwent colonoscopy after a positive fecal occult blood test. The colonoscopist found a 20-mm pedunculated polyp in the descending colon and performed endoscopic resection. Histopathologic examination revealed non-solid type poorly differentiated adenocarcinoma. The lesion invaded the submucosa (3500 μm from the muscularis mucosa) and demonstrated lymphatic invasion. In spite of the early stage of this cancer, the patient was considered at high risk for nodal metastasis. She was referred to our institution, where she underwent bowel resection. Although there was no residual cancer after her endoscopic resection, a metastatic lesion was found in one regional lymph node. The patient is undergoing postoperative adjuvant chemotherapy, and there has been no evidence of recurrence 3 months after the second surgery. Conclusions Additional bowel resection is indicated for patients with pedunculated polyps and multiple risk factors for nodal metastasis, such as poorly differentiated adenocarcinoma and lymphatic invasion. We encountered just such a patient who did have a nodal metastasis; herein, we report her case history with a review of the literature.

Abstract Background Metastasis to the breast is rare. We herein report a patient with metastatic medullary thyroid carcinoma to the breast for whom measuring the calcitonin level was an important clue to the correct diagnosis. Case presentation A 54-year-old woman visited our hospital for the treatment of recurrent metastatic medullary thyroid carcinoma due to multiple endocrine neoplasia 2A and breast cancer. Positron emission tomography performed before the operation for metastatic medullary thyroid carcinoma recurrence in the neck showed the accumulation of 18F-fluorodeoxyglucose in the bilateral breast at sites other than the disease in the neck. Ultrasonography revealed multiple tumors in both breasts. A core needle biopsy of three breast tumors was performed. Microscopically, the tumor cells showed solid growth and did not show a tubular structure. She was diagnosed with triple-negative invasive ductal carcinoma. Post-operative positron emission tomography was performed as the serum calcitonin level increased after the operation. The accumulation of 18F-fluorodeoxyglucose in the bilateral breast tumors and lymph nodes in the neck was noted. The possibility of the breast tumors being metastasis of metastatic medullary thyroid carcinoma was considered. Needle aspiration was performed for three breast tumors. The calcitonin level of the washout fluid was measured and found to be ≥ 17,500 pg/mL. Immunohistochemistry showed that the tumor cells were calcitonin-positive and gross cystic disease fluid protein-15-negative. Vandetanib was started as recurrent metastatic medullary thyroid carcinoma with breast metastasis was finally diagnosed. The serum calcitonin level decreased after 1 month. Conclusion Although breast metastasis of medullary thyroid carcinoma is rare, a correct diagnosis is indispensable for appropriate treatment. When a breast tumor shows atypical morphological features for breast cancer according to the histopathology in a patient with a history of cancer, metastasis to the breast should be considered. Calcitonin measurement of the needle washout fluid was useful for confirming metastatic medullary thyroid carcinoma.

Abstract Long-term patterns in trajectories of natural communities provide insights into ecological resilience, but their assessment requires long-term census data. We analyzed 16-year census data for intertidal communities from 30 rocky shores along Japan’s Pacific coast to assign community change to four possible trajectories (stable, reversible, abrupt, or linear) representing different aspects of ecological resilience, and to estimate multiple metrics of temporal invariability (species richness, species composition, and community abundance). We examined (1) how the prevalence of the four trajectories differs among regions, (2) how the features (model coefficients) of each trajectory vary among regions, and (3) how the temporal invariabilities differ among trajectories and regions. We found that the stable trajectory was the most common. Its features differed among regions, with a faster recovery to steady-state equilibrium in low-latitude regions. Furthermore, trajectories and temporal invariabilities both varied among regions, seemingly in association with the strength of ocean current fluctuations. Thus, the relationship between community temporal invariability and trajectory may be weak or absent, at least at the regional scale.

Key message Pulmonary amyloidosis should be included in the differential diagnosis of calcified lung nodules, and more careful preparation for bleeding should be taken when performing bronchoscopy. While management does not require aggressive treatment, follow‐up is necessary to monitor for multiple myeloma and malignant lymphoma

Acute distress immediately following an 18F-fluorodeoxyglucose positron emission tomography/computed tomography (FDG-PET/CT) scan is an exceedingly rare event. We report a case whose condition was suddenly deteriorated in the nuclear medicine laboratory, and whose diagnosis was confirmed by FDG-PET/CT. A 67-year-old woman with left renal cell carcinoma (RCC) suddenly complained of dyspnea and tachycardia just after undergoing FDG-PET/CT. PET/CT images showed increased FDG uptakes in the left renal vein, inferior vena cava, right atrium, and bilateral hila. She was diagnosed with a massive tumor embolism from the inferior vena cava to both pulmonary arteries, and urgently underwent tumor embolectomy. FDG-PET/CT was helpful for diagnosing the tumor embolism and differentiating it from bland thromboembolism in this patient with RCC.

Abstract We report a rare case of ovarian carcinoma in which a mediastinal recurrence was resected 16 years after the initial operation. A 72‐year‐old woman underwent hysterectomy with adnexectomy for stage IIIC ovarian serous carcinoma after neoadjuvant chemotherapy. Six courses of adjuvant chemotherapy were administered. Three years after surgery, left supraclavicular lymph node metastasis occurred, and radiotherapy and two courses of chemotherapy were administered. Six years before presentation, a metastasis at the right cardiophrenic lymph node was resected, and six courses of chemotherapy were administered. During follow‐up, a retrosternal tumour was found. The metastatic lesion in contact with the diaphragm was thought to result from pleuroperitoneal communication, and it increased in size. Although high‐grade serous carcinoma is aggressive, its sensitivity to chemotherapy may suppress early recurrence, contributing to good outcomes, but with late recurrence. Multidisciplinary therapy including surgery is required for improved long‐term prognosis for mediastinal metastasis of ovarian serous carcinoma.

Abstract Background Parathyroid carcinoma as a cause of primary hyperparathyroidism in children is extremely rare. We report a case of parathyroid carcinoma which occurred in a 13-year-old girl who survived for more than 45 years after the first operation. Case presentation A woman was admitted to our hospital for the treatment of recurrent parathyroid carcinoma in the neck. She had been diagnosed with primary hyperparathyroidism from a fibula fracture and underwent parathyroidectomy at 13 years old. She had no family history of multiple endocrine neoplasia or jaw tumor syndrome. Genetic testing was not performed, and the histopathological diagnosis of the tumor had been parathyroid adenoma at the time. At 22 years old, she showed hypercalcemia after a femur fracture. Pulmonary metastases of parathyroid carcinoma in the bilateral lungs were found and surgically removed. Regarding the clinical course, her diagnosis was corrected from parathyroid adenoma to parathyroid carcinoma. At 33 years old, re-resection of the lung metastases was performed. For 10 years, her serum calcium level stayed within the normal range. However, her serum calcium level and intact parathyroid hormone eventually began to increase. Two masses suspected of being parathyroid carcinoma recurrence were found in the neck when she was 57 years old. En bloc resection was performed. Pathologically, the tumors were diagnosed as parathyroid adenoma. The serum calcium level and intact parathyroid hormone did not decrease after the operation. A 99mTc-methoxy-isobutyl-isonitrile- and 18F-fluorodeoxyglucose-negative, 11C-methionine-positive tumor was detected at the right side of the trachea in the neck. The tumor was removed, along with the thyroid, muscle, and trachea that were involved. The pathological diagnosis was parathyroid carcinoma recurrence. The serum calcium level and intact parathyroid hormone decreased temporarily but had increased again 8 months later. Methionine-positive tumors were found at the right side of the trachea and suspected of being a recurrence. Denosumab reduced her serum calcium level, and radiation successfully suppressed the growth of the recurrent tumors. Conclusion We have reported a rare case of parathyroid carcinoma in a child who has survived for over 40 years. Positron emission tomography of 11C-methionine was useful for detecting local recurrence. This patient’s long-term survival has been attributed to multimodality treatment including repeated surgery, medication, and radiation.

Novel coronavirus (SARS-Coronavirus-2:SARS-CoV-2) which emerged in Wuhan, China, has spread to multiple countries rapidly. We report the first case of meningitis associated with SARS-CoV-2 who was brought in by ambulance due to a convulsion accompanied by unconsciousness. He had never been to any foreign countries. He felt generalized fatigue and fever (day 1). He saw doctors nearby twice (day 2 and 5) and was prescribed Laninamivir and antipyretic agents, His family visited his home and found that he was unconsciousness and lying on the floor in his vomit. He was immediately transported to this hospital by ambulance (day 9). Under emergency transport, he had transient generalized seizures that lasted about a minute. He had obvious neck stiffness. The specific SARS-CoV-2 RNA was not detected in the nasopharyngeal swab but was detected in a CSF. Anti- HSV 1 and varicella-zoster IgM antibodies were not detected in serum samples. A brain MRI showed hyperintensity along the wall of right lateral ventricle and hyperintense signal changes in the right mesial temporal lobe and hippocampus, suggesting the possibility of SARS-CoV-2 meningitis. This case warns the physicians of patients who have CNS symptoms.

Abstract Background Robot-assisted thoracoscopic surgery (RATS) is useful for surgery in the apical region of the chest cavity, as it narrows towards the head. Here, we describe a nonfunctional, rib-invasive paraganglioma arising in the posterior mediastinum that was successfully removed using RATS combined with chest wall resection. Case presentation A 31-year-old woman presented with a posterior mediastinal mass on chest computed tomography (CT) scan during a medical check-up 2 years prior. Positron emission tomography/computed tomography scan with F-18 fluorodeoxyglucose revealed a mass associated with standardized uptake maximum value of 2.69. With a preoperative diagnosis of neurogenic tumor by CT-guided percutaneous fine-needle aspiration biopsy, we performed robot-assisted tumor resection combined with chest wall resection. The wristed instruments of the robotic surgical system have increased range of motion and enabled the tumor resection without organ injury in the thoracic cavity. Histopathology examination revealed a non-functional paraganglioma with rib invasion. Conclusions RATS is a useful technique, enabling safer and easier resection of a mediastinal tumor adjacent to surrounding organs.

Abstract Our report presents the smallest bronchial carcinoid thus far associated with peripheral squamous cell carcinoma. A 79‐year‐old Japanese man presented with an abnormal shadow on chest radiography. Chest computed tomography revealed an ill‐defined nodule at the left S9a area without lymph node involvement. At the time of bronchoscopy, a nodule of ~1 mm in diameter with a smooth surface was observed at the orifice of the left B8. Left lower lobectomy was performed. Pathologically, the S9a tumour was keratinizing squamous cell carcinoma with surgical stage IA3, and the left B8 tumour was a typical bronchial carcinoid. The patient was alive without recurrence 2 years after the surgery. Cases of combined carcinoid and lung cancer are rare. It may be possible to identify bronchial carcinoids early by careful observation of the bronchi during bronchoscopy for peripheral neoplastic lesions.

Relapse is a limitation for the efficacy of the anaplastic lymphoma kinase (ALK)-inhibitor alectinib in ALK-rearranged lung cancer. Here, the authors show that YAP1 activation upon alectinib treatment leads to therapy resistance and that inhibiting both YAP1 and ALK leads to longer tumor remission in mice.

Primary aldosteronism (PA) is more common than expected. Aberrant adrenal expression of luteinizing hormone (LH) receptor in patients with PA has been reported; however, its physiological role on the development of PA is still unknown. Herein, we report two unique cases of PA in patients with untreated Klinefelter’s syndrome, characterized as increased serum LH, suggesting a possible contribution of the syndrome to PA development. Case 1 was a 39-year-old man with obesity and hypertension since his 20s. His plasma aldosterone concentration (PAC) and renin activity (PRA) were 220 pg/mL and 0.4 ng/mL/h, respectively. He was diagnosed as having bilateral PA by confirmatory tests and adrenal venous sampling (AVS). Klinefelter’s syndrome was suspected as he showed gynecomastia and small testes, and it was confirmed on the basis of a low serum total testosterone level (57.3 ng/dL), high serum LH level (50.9 mIU/mL), and chromosome analysis. Case 2 was a 28-year-old man who had untreated Klinefelter’s syndrome diagnosed in his childhood and a 2-year history of hypertension and hypokalemia. PAC and PRA were 247 pg/mL and 0.3 ng/mL/h, respectively. He was diagnosed as having a 10 mm-sized aldosterone-producing adenoma (APA) by AVS. In the APA, immunohistochemical analysis showed co-expression of LH receptor and CYP11B2. Our cases of untreated Klinefelter’s syndrome complicated with PA suggest that increased serum LH levels and adipose tissues, caused by primary hypogonadism, could contribute to PA development. The possible complication of PA in hypertensive patients with Klinefelter’s syndrome should be carefully considered.

Meigs' syndrome (MS) is defined as a benign ovarian tumor with pleural effusion and ascites, which, resolve after its removal. The pathogenesis of ascites and pleural effusion in MS is unknown. A 64-year-old woman with a history of uterine fibroid was admitted owing to dyspnea over 1 month. A chest radiograph showed right hydrothorax. Carbohydrate antigen 125 (CA-125) level was 645 U/mL. Computed tomography of chest, abdominal, and pelvis showed hydrothorax in the right thoracic cavity and atelectasis of the right lower lobe, a tumor, 14 cm in diameter, and ascites in the pelvis. Despite chest drainage for hydrothorax, the amount of pleural effusion was over 300 ml/days every day. Pleural fluid was a lymphocyte-predominant exudate. The bacterial mycobacterial cultures of the pleural fluid were negative. Cytological examination of the pleural fluid showed no malignancy. As the combination of pleural effusion, ascites, and ovarian tumor was suggestive of MS, the patient was referred to the Department of Gynecology. She underwent total hysterectomy and bilateral salpingo-oophorectomy. Histopathological examination of the resected tumor confirmed fibrothecoma. Thus, it is important to consider the possibility of MS in female patients with unexplained pleural effusion and an ovarian tumor.

Background: Gastric leiomyomas are usually solitary, rounded, and demarcated masses, and multiple leiomyomas (more than three) are extremely rare. We report a case of a 30-year-old woman who was referred to our hospital for further treatment of an enlarging gastric submucosal tumour (SMT).Case Presentation: She underwent laparoscopic gastric partial resection with oesophagogastroduodenoscopy (OGD), which revealed a multinodular gastric SMT (10 cm in diameter) located in the upper body of the stomach. Endoscopic ultrasonography (EUS) revealed a hypoechoic mass located in the fourth layer and multiple nodules approximately 1 cm in diameter. We subsequently performed EUS-guided fine-needle aspiration biopsy. The patient underwent laparoscopic gastric partial resection with EGD. Because the tumour was extensive on the lesser curvature of the stomach and was located near the oesophagogastric junction, we preserved the gastric function with minimal resection of the stomach. No intraoperative or postoperative complications were observed. The tumour was pathologically diagnosed as gastric leiomyoma. Laparoscopic minimized resection used by endoscope, can avoid gastric deformity and preserve gastric function, especially when the gastric SMTs are located near the oesophagogastric junction.Conclusion: Laparoscopic gastric partial resection with EGD is an effective treatment option for complete resection of extensive gastric SMT.

Abstract Primary tumours establish metastases by interfering with distinct organs. In pre‐metastatic organs, a tumour‐friendly microenvironment supports metastatic cells and is prepared by many factors including tissue resident cells, bone marrow‐derived cells and abundant fibrinogen depositions. However, other components are unclear. Here, we show that a third organ, originally regarded as a bystander, plays an important role in metastasis by directly affecting the pre‐metastatic soil. In our model system, the liver participated in lung metastasis as a leucocyte supplier. These liver‐derived leucocytes displayed liver‐like characteristics and, thus, were designated hepato‐entrained leucocytes (HepELs). HepELs had high expression levels of coagulation factor X (FX) and vitronectin (Vtn) and relocated to fibrinogen‐rich hyperpermeable regions in pre‐metastatic lungs; the cells then switched their expression from Vtn to thrombospondin, both of which were fibrinogen‐binding proteins. Cell surface marker analysis revealed that HepELs contained B220+CD11c+NK1.1+ cells. In addition, an injection of B220+CD11c+NK1.1+ cells successfully eliminated fibrinogen depositions in pre‐metastatic lungs via FX. Moreover, B220+CD11c+NK1.1+ cells demonstrated anti‐metastatic tumour ability with IFNγ induction. These findings indicate that liver‐primed B220+CD11c+NK1.1+ cells suppress lung metastasis.

Objective: Atypical polypoid adenomyoma (APAM) is an epithelial-mesenchymal mixed tumor which often develops in the uterine cavity of reproductive age women, requiring preservation of the reproductive functions. Preoperative endometrial biopsy may not yield histological diagnosis as the tumor is a solid smooth muscle tumor. The standard treatment option is a hysteroscopic resection for the diagnosis and the treatment at the same time. Case report: We report a case of rapidly-growing APAM successfully diagnosed preoperatively via transcervical punch biopsy followed by a laparoscopic resection. The mass was relatively large, had been located in the lower segment of the uterus, and the area of contact with the muscular layers was large. It was a complete removal and no recurrence had been observed 9 months after the operation. Conclusion: This is the first report of APAM treated by laparoscopic resection. The method may be a useful alternative when hysteroscopic surgery is inappropriate.

Objectives: To assess the predictive values of myenteric and submucosal plexitis for postoperative endoscopic recurrence of Crohn's disease (CD). Methods: A retrospective study of CD patients who underwent intestinal resection between 1995 and 2013 in the Department of Surgery 2, Tokyo Women's Medical University was performed. Proximal resection margins were analyzed and plexitis was evaluated by counting the number of inflammatory cells in myenteric and submucosal plexuses. The sizes of the most severely inflamed ganglion (MIG) were measured. Multiple regression analysis was used to identify independent risk factors for postoperative endoscopic recurrence. Results: Of the 51 included patients, 40 patients underwent colonoscopy after surgery. Endoscopic recurrence was observed in 21 patients (52.5%). Mean duration (±standard deviation) from surgery to recurrence was 49.7±34.7 months. Endoscopic recurrence rates at 1, 3, and 5 years were 5.0%, 24.1%, and 45.1%, respectively. Submucosal plexitis and myenteric plexitis were observed in 36 (90.0%) and 37 patients (92.5%), respectively. On multivariate analysis, initial intestinal resection, rate of plexitis

In primary infection with Salmonella, it has been reported—without consideration of Salmonella's functions—that humoral immunity plays no role in the clearance of bacteria. In fact, Salmonella targets and suppresses several aspects of humoral immunity, including B cell lymphopoiesis, B cell activation, and IgG production. In particular, the suppression of IgG-secreting plasma cell maintenance allows the persistence of Salmonella in tissues. Therefore, the critical role(s) of humoral immunity in the response to Salmonella infection, especially at the late phase, should be re-investigated. The suppression of IgG plasma cell memory strongly hinders vaccine development against non-typhoidal Salmonella (NTS) because Salmonella can also reduce humoral immune memory against other bacteria and viruses, obtained from previous vaccination or infection. We propose a new vaccine against Salmonella that would not impair humoral immunity, and which could also be used as a treatment for antibody-dependent autoimmune diseases to deplete pathogenic long-lived plasma cells, by utilizing the Salmonella's own suppression mechanism of humoral immunity.

Summary:. Hemangioma in the submandibular gland or submandibular space is rare. A 31-year-old woman was referred to our department. She became aware of painless swelling in the right submandibular gland at 9 months ago. The right submandibular gland showed swelling with no tenderness, and the palpation was softer than the normal submandibular gland. Magnetic resonance imaging short T1 inversion recovery (STIR) sequence showed a 38 × 29 mm well-circumscribed, lobulated mass with high signal intensity attached with the submandibular gland. Computed tomography showed phlebolith formation. A clinical diagnosis of suspected hemangioma in submandibular gland was made, and submandibular gland excision including mass was performed with submandibular approach without complications. No unusual bleeding during the surgery was observed. Pathological finding indicated that thick walled vessels containing smooth muscle were proliferated with dilatation. Some vascular lumen included phlebolith calcification. Although the boundary with the submandibular gland was mainly clear, some parts were irregularly occupied with a component of hemangioma. The final diagnosis was venous hemangioma in submandibular gland. No postoperative recurrence was recognized in 1-year follow-up. A search of PubMed revealed that only 18 cases associated with hemangioma in the submandibular gland in 12 English articles. Literature review indicated that (1) approximately 70% patients showed painful swelling and (2) x-ray or computed tomography may indicate the phlebolith calcification. It may make an erroneous diagnosis of sialolithiasis. Magnetic resonance imaging finding would be needed for the diagnosis.

This paper discusses school safety mainly in relation to safety education and examines measures for the comprehensive development of systems to ensure the safety of children at school, including those requiring medical care. The number of children requiring medical care is increasing in general schools following the promotion of inclusive education. The government of Japan has established the School Health and Safety Act and Guidelines on School Curricula. Municipalities have developed education systems that cover the safety education specified in disaster preparedness plans for schools. Safety education has been promoted through problem-oriented and experience-based methods as well as other methods of learning to date. Considering the outcomes of these systems and approaches, it is expected that safety management systems in schools, especially general schools that include children requiring medical care, will also develop in combination with safety education.

Objectives: Adenocarcinoma is known to be associated with ulcerative colitis, but the diagnosis is sometimes challenging, both clinically and pathologically. Methods and Results: We present a case of extremely well-differentiated adenocarcinoma associated with ulcerative colitis, in which preoperative diagnosis was not possible. Glands in biopsy specimens showed a serrated appearance that looked like low-grade dysplasia or regenerative mucosa. After an operation due to severe symptoms of stenosis, carcinoma was diagnosed. Tumor cells, especially in invasive glands, tended to show stronger immunoreactivity against anti-CK7, TNF-α and Aurora B antibodies compared to cells of mucosal lesion. Interestingly, CD44v6, one of the adhesion molecules, was less expressed in invasive glands, while those glands exhibited stronger expression of a disintegrin and metalloproteinase 17 (ADAM 17), one of the sheddases that cleaves an extracellular domain of CD44. Conclusions: These observations appear interesting to consider the pathogenesis and to diagnose extremely well-differentiated adenocarcinoma in ulcerative colitis, although further investigation is needed.

Based on the results of our surveys involving several municipal departments of education that support the enrollment of children requiring medical care in general schools, this report describes Japanese systems to support inclusive education for these children, the current status of such support, and related challenges. The municipal departments of education that systematically support inclusive education for children requiring medical care began to promote such education in their communities before the initiation of nationwide enrollment support for the children. In addition, their sections in charge of enrollment support also manage affairs related to human rights education, rather than general affairs for special support and education. Such a variation in the sections in charge resulted in differences in the purposes of support. Further nationwide surveys should be conducted to develop universal design principles and comprehensively support inclusive education for children requiring medical care.

Early childhood education and care (ECEC) workers and hospital play specialists (HPSs) share a role in supporting the development of sick children through play while respecting their autonomy. On the other hand, in supporting children’s play, managing their anxiety, and making environmental arrangements for them, ECEC workers and HPSs play different roles. When supporting the development of sick children, the former should respect their autonomy and make the most of the characteristics of ECEC as a measure to comprehensively support their participation in play, including it in their daily lives. ECEC workers are also expected to contribute to the further development of comprehensive support by promoting collaboration and cooperation with various professionals, including HPSs.