{"title":"Neuromyelitis Optica Spectrum Disorder with Interstitial Pneumonia and Marked HyperCKemia as an Initial Presentation: A Case Report and Literature Review.","authors":"Tomohiro Yoshida, Keisuke Nishimura, Kei Hirose, Hiroyuki Murabe","doi":"10.1620/tjem.2025.J007","DOIUrl":"10.1620/tjem.2025.J007","url":null,"abstract":"<p><p>Neuromyelitis optica spectrum disorder is an inflammatory disease of central nervous system. Autoantibodies against aquaporin 4 (anti-AQP4-ab) are often present in neuromyelitis optica spectrum disorder and have pathogenic effects on central nervous system. Aquaporin 4 is also expressed in peripheral organs, and some cases of anti-AQP4-ab-positive neuromyelitis optica spectrum disorder with peripheral organ involvement have recently been reported. Here, we report a unique case of anti-AQP4-ab-positive neuromyelitis optica spectrum disorder. A 58-year-old man was admitted with fever, interstitial pneumonia, and markedly increased serum-creatine kinase levels (up to 300,200 U/L). Skeletal muscle biopsy revealed rhabdomyolysis. Collagen and infectious diseases were excluded; therefore, adverse effects of prescription drugs were initially suspected. Clinical symptoms improved with steroid administration and discontinuation of all previously prescribed medications. However, bilateral vision loss, bilateral lower-limb muscle weakness, and bilateral lower-limb sensory disturbances appeared later, and fever and increased serum-creatine kinase levels recurred. The patient was diagnosed with neuromyelitis optica spectrum disorder with peripheral organ (lung and skeletal muscles) involvement based on magnetic resonance imaging findings and serum anti-AQP4-ab positivity. Our case demonstrates that interstitial pneumonia and rhabdomyolysis can be the initial symptoms and indicators of the relapse of anti-AQP4-ab-positive neuromyelitis optica spectrum disorder, and skeletal muscle involvement can lead to markedly elevated creatine kinase levels.</p>","PeriodicalId":23187,"journal":{"name":"Tohoku Journal of Experimental Medicine","volume":" ","pages":"327-334"},"PeriodicalIF":1.6,"publicationDate":"2025-08-07","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143067785","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
{"title":"Inhibition of Regulator of G Protein Signaling 20 Reduces Cell Proliferation, Invasion and Migration by Activating the Hippo Pathway in Cutaneous Melanoma.","authors":"Yi Zhang, Mengbi Gong","doi":"10.1620/tjem.2025.J097","DOIUrl":"https://doi.org/10.1620/tjem.2025.J097","url":null,"abstract":"","PeriodicalId":23187,"journal":{"name":"Tohoku Journal of Experimental Medicine","volume":" ","pages":""},"PeriodicalIF":1.6,"publicationDate":"2025-07-31","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"144754383","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Naoki Tokunaga, Hisato Shima, Takuya Okamoto, Masato Maekawa, Jun Minakuchi
{"title":"The Far Negative Intercept of the Regression Formula Obtained from Repeated Measurements of BChE and Albumin Levels is Useful for Detecting Hereditary Heterozygous Butyrylcholinesterase Deficiency in Patients on Dialysis.","authors":"Naoki Tokunaga, Hisato Shima, Takuya Okamoto, Masato Maekawa, Jun Minakuchi","doi":"10.1620/tjem.2024.J149","DOIUrl":"10.1620/tjem.2024.J149","url":null,"abstract":"<p><p>Serum butyrylcholinesterase (BChE) levels are positively correlated with serum albumin levels and are influenced by inflammation. Heterozygous BChE deficiency in patients on dialysis may be overlooked as malnutrition owing to dialysis-associated low BChE and albumin levels. In this study, we aimed to clarify a method for identifying low BChE levels due to hereditary causes. This single-center, retrospective, observational study included 1,104 patients undergoing dialysis and 1,716 patients not undergoing dialysis. Patients on dialysis with available data of high-sensitivity C-reactive protein (hsCRP) levels were divided into three groups according to the hsCRP level: low (< 1 mg/L), average (1-3 mg/L), and high (> 3 mg/L), and the association of serum hsCRP level with BChE and albumin levels was investigated. We compared intercept values (y value at x = 0) of the regression formula obtained from repeated measurements between patients on dialysis and those with a known hereditary heterozygous BChE deficiency. Serum BChE and albumin levels in patients on dialysis were significantly lower, and a positive correlation was observed. The hsCRP level and the BChE and albumin levels were negatively correlated. BChE and albumin levels were highest in the low hsCRP group and lowest in the high hsCRP group, and a significant positive correlation was observed in all groups. The intercept values of the regression formula obtained from repeated measurements of BChE and albumin levels in patients on dialysis and a patient with hereditary heterozygous BChE deficiency were 0.383 and ---102.730, respectively, which may be useful in distinguishing hereditary causes.</p>","PeriodicalId":23187,"journal":{"name":"Tohoku Journal of Experimental Medicine","volume":" ","pages":"311-317"},"PeriodicalIF":1.6,"publicationDate":"2025-07-30","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143012242","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
{"title":"Cancer-Associated Fibroblasts in Gallbladder Cancer Modify the Migration and Invasion of Gallbladder Cancer Cells.","authors":"Kazuhiko Tsukuda, Masaharu Ishida, Takayuki Miura, Shimpei Maeda, Hideo Ohtsuka, Masamichi Mizuma, Kei Nakagawa, Michiaki Unno","doi":"10.1620/tjem.2024.J126","DOIUrl":"10.1620/tjem.2024.J126","url":null,"abstract":"<p><p>The importance of the microenvironment in cancer progression is widely recognized, and interactions between cancer cells and stromal cells play an important role in the progression of the disease. A major component of stromal cells are fibroblasts, known as cancer-associated fibroblasts (CAFs). CAFs are thought to enhance the malignant properties of cancer cells through various secreted proteins. It is also known that CAFs function as a leading cell in cancer invasion, and their migratory ability is involved in local cancer invasion. The aim of this study was to elucidate the function of CAF in gallbladder cancer, which is one of the gastrointestinal malignancies with the worst prognosis.CAFs were primarily cultured from surgical specimens of gallbladder cancer patients. We investigated the migration ability of established CAFs and the effects of conditioned medium obtained from CAFs on the growth and invasion ability of gallbladder cancer cell lines. Functional analysis showed that the migration ability of certain CAFs was enhanced compared to control, and that conditioned medium enhanced the migration and invasion of gallbladder cancer cell lines. Gene expression analysis of CAFs revealed that tenascin-C (TNC) and podoplanin (PDPN) were highly expressed in CAFs with the enhancing functions. Immunohistochemical staining of TNC and PDPN on surgical specimens was performed to investigate the relationship with the prognosis. Disease-free survival and overall survival were found to be reduced in patients with high expression of those genes. The results of this study indicate that CAFs expressing TNC and PDPN promote cancer progression in gallbladder cancer.</p>","PeriodicalId":23187,"journal":{"name":"Tohoku Journal of Experimental Medicine","volume":" ","pages":"279-293"},"PeriodicalIF":1.7,"publicationDate":"2025-07-17","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142591465","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
{"title":"Endoscopic Surgery for Facial Subcutaneous and Nasal Submucosal Abscesses in a Child.","authors":"Tomotaka Hemmi, Shuji Chiba, Kazuhiro Nomura","doi":"10.1620/tjem.2025.J016","DOIUrl":"10.1620/tjem.2025.J016","url":null,"abstract":"<p><p>We herein report a case of abscess formation from the subcutaneous area of the face to the submucosa of the nasal cavity. To our knowledge, this case was unprecedented, but we were able to cure the patient by applying endoscopic sinus surgery techniques. A 13-year-old boy visited a local clinic because of right facial swelling. Contrast-enhanced computed tomography (CT) showed abscess formation from the subcutaneous region of the face to the submucosa of the nasal cavity, and endoscopic surgery was performed. In surgery, endoscopic modified medial maxillectomy (EMMM) and a direct approach to the anterior and lateral parts of the maxillary sinus with an endoscope (DALMA) were applied to perform radical drainage. The patient was discharged from the hospital on postoperative day 7 after completion of antimicrobial therapy because of good progress. Two months postoperatively, healing of the incision and maintenance of openness of the maxillary sinus were confirmed, and the follow-up was terminated. Endoscopic surgery is the first-choice treatment because of its superior therapeutic efficacy and functional preservation.</p>","PeriodicalId":23187,"journal":{"name":"Tohoku Journal of Experimental Medicine","volume":" ","pages":"273-277"},"PeriodicalIF":1.7,"publicationDate":"2025-07-17","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"143410858","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
{"title":"Development of a Temporary Custody Re-Entry Risk Prediction Model using Longitudinal Cohort Data in Japan.","authors":"Sotaro Tanaka, Inoue Mariko, Yamaoka Kazue","doi":"10.1620/tjem.2024.J144","DOIUrl":"10.1620/tjem.2024.J144","url":null,"abstract":"<p><p>The number of temporary custody cases due to child maltreatment and other reasons at child guidance centers in Japan is increasing, prompting imperative scrutiny of re-entry risk assessment. We aimed to derive and internally validate a risk prediction model for re-entry into temporary custody. The risk prediction model was developed from an analysis of data from 725 children recruited in this retrospective single-center longitudinal cohort study conducted in Japan. The anticipated outcome was re-entry into temporary custody. Predictor variables were selected from 15 prospective variables concerning information on children and their familial contexts. A risk prediction model was developed using stepwise logistic regression. The final risk model was validated via C-statistic using cross-validation and bootstrap resampling methods. Calibration was assessed using the Hosmer-Lemeshow test. Of 725 children under temporary custody, 178 (24.6%) experienced re-entry into temporary custody. The predictors in the conclusive risk model were a history of temporary custody (p < 0.001), age at first birth of < 25 years (p < 0.002), single mother or stepfamily (p < 0.001), reasons for temporary custody including child abuse (p = 0.076) and child sex (p = 0.152), and child disability certificate (p = 0.252). Calibration scrutiny via the Hosmer-Lemeshow test revealed no discernible irregularities (p = 0.320). The naïve C-statistic of the model was 0.70, whereas the optimism-corrected C-statistics was 0.67-0.69. The presented risk prediction model showed acceptable calibration and discriminatory capability. The model can optimize limited human resources by providing valid risk estimates of the likelihood of re-entry to temporary custody within one year.</p>","PeriodicalId":23187,"journal":{"name":"Tohoku Journal of Experimental Medicine","volume":" ","pages":"295-303"},"PeriodicalIF":1.7,"publicationDate":"2025-07-17","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142814204","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}