Background Cardiac surgery in late pregnancy requires balancing maternal rescue against fetal risk. Case Summary A 31-week pregnant woman with prior resection of a left atrial “myxoma” presented with acute heart failure caused by recurrent masses prolapsing through the mitral valve. Because the fetus was viable and maternal collapse was imminent, a 7-F, 35-mL intra-aortic balloon pump was inserted, cesarean delivery was performed, and redo open-heart surgery followed the next day. A 1,638-g male infant with Apgar scores of 7 and 8 was delivered. Two posterior left atrial tumors were resected; one involved the mitral leaflet and required mechanical mitral valve replacement. Re-review of the recurrent and original specimens showed morphology and immunophenotype consistent with myxofibrosarcoma. Discussion Staged delivery before cardiopulmonary bypass can protect a viable fetus in hemodynamically critical maternal disease. Take-Home Message Pathological re-evaluation is essential when “recurrent myxoma” behaves atypically.
A 52-year-old female patient was initially evaluated at another hospital for a splenic tumor. Chest computed tomography revealed a 12-mm nodule with well-defined margins and uniform internal density in the S5 segment of the right middle lobe. She was referred to our hospital for follow-up of a suspected benign lung tumor. After 9 months of outpatient observation, the nodule increased in size to 16 mm, prompting referral to our department. Thoracoscopic partial resection of the right lung was performed for both diagnostic and therapeutic purposes. Histopathological examination showed a tumor composed of densely packed spindle-shaped cells with eosinophilic cytoplasm and minimal atypia. The tumor was positive for estrogen receptor and progesterone receptor. Considering the patient' s history of uterine myoma, these findings led to a diagnosis of benign metastasizing leiomyoma (BML). BML is a rare condition in which benign uterine leiomyomas metastasize to the lungs. It should be considered in the differential diagnosis of women with a history of uterine myoma. Although BML typically presents with multiple pulmonary lesions, solitary lesions are rare. The present case, with a single lesion, posed a challenge in preoperative diagnosis. Therefore, we report this case along with a literature review.
Cardiac hemangiomas located in the right heart can lead to serious complications, including right ventricular outflow tract (RVOT) obstruction, arrhythmias, and sudden cardiac death. Although surgical resection remains the primary treatment, complete excision of intramural tumors poses risk of impairing cardiac function. This case report describes the successful resection of a large right ventricular hemangioma using intraoperative voltage mapping, which enable maximal tumor removal while preserving myocardial integrity and preventing postoperative heart failure. A 76-year-old female underwent a routine health examination in July 2024, during which cardiomegaly and nonspecific ST-segment changes were detected on electrocardiography. Contrast enhanced computed tomography revealed a well-defined 60 mm mass within the right ventricle, causing significant ROVT stenosis. No evidence of distant metastasis or elevated tumor markers was detected. The patient underwent tumor resection via median sternotomy. Intraoperative voltage mapping was utilized to delineate viable myocardium at the tumor margins. The tumor was excised while preserving functional myocardial tissue. Cryoablation was performed at the resection margins, and resultant defect in the right ventricular wall was reconstructed using a bovine pericardial patch. Histopathological analysis confirmed the diagnosis of cardiac hemangioma. The patient experienced an uneventful postoperative course had no postoperative complications and was discharged on postoperative day 16. Preoperative and postoperative cardiac magnetic resonance imaging demonstrated preserved right ventricular function. Intraoperative voltage mapping proved to be a valuable adjunct in the surgical management of right ventricular tumors, enabling effective tumor resection while preserving myocardial tissue and maintaining postoperative cardiac function.
We herein report an autopsy case of streptococcal toxic shock syndrome (STSS) with disseminated intravascular coagulation and multiple cerebral infarctions induced by Streptococcus dysgalactiae subsp. equisimilis in an 84-year-old male. Pathological examination revealed sepsis with hemophagocytosis in the reticular system and intravascular bacteria in multiple organs, originating from bacterial necrotizing fasciitis of the lower extremities. The brain MRI findings showed a diffusion weighted imaging-fluid-attenuated inversion-recovery mismatch, whereas the pathology was almost normal, thus supporting a hyperacute phase of cerebral infarction. The findings in this case help to elucidate the pathogenesis of STSS and develop appropriate treatment strategies.
Introduction:A previous report has shown that cyclooxygenase-2 inhibitors can prevent the recurrence of cystitis glandularis postoperatively. Herein, we present a case of cystitis glandularis in which the tumor volume was markedly reduced by preoperative oral administration of a cyclooxygenase-2 inhibitor. Case presentation:A 45-year-old man with voiding difficulty and lower abdominal pain during urination was referred to our hospital. Cystoscopy revealed multiple cystitis glandularis-like edematous masses on the trigone and the neck of the bladder, completely involving the bilateral ureteral orifices. Cyclooxygenase-2 inhibitor was orally administered at the patient's request. Six weeks later, the tumor volume was markedly reduced, bilateral ureteral orifices were identified, and the voiding difficulty and pain on urination disappeared. Complete transurethral resection of the residual tumor was performed, and the pathological diagnosis was intestinal-type cystitis glandularis. Conclusion:Cyclooxygenase-2 inhibition can be considered a useful therapeutic strategy for cystitis glandularis.
Mixed connective tissue disease (MCTD) is characterized by mixed features of systemic lupus erythematosus, systemic sclerosis, and polymyositis/dermatomyositis and is rare in children. Here, we report a case of MCTD in a 10-year-old girl who presented at our hospital with arthralgia, Raynaud's phenomenon, and fatigue. Blood tests were positive for anti-U1-ribonucleoprotein (RNP) antibodies and for rheumatoid factors (RFs) IgG-RF and anti-galactose-deficient IgG. Levels of myogenic enzymes and hypergammaglobulinemia were elevated. Macrophages were prominent in bone marrow, with scattered phagocytic macrophages. MCTD was diagnosed based on the patient's symptoms and laboratory findings. Methylprednisolone pulse therapy combined with oral tacrolimus was administered, which led to resolution of symptoms. Three months after pulse therapy, arthralgia worsened and methotrexate was administered. Arthralgia improved but did not resolve. Magnetic resonance imaging performed to investigate the hip pain revealed a mature ovarian teratoma, which was surgically removed. Because the pain persisted and interfered with her daily life, she was treated with tocilizumab for joint pain relief, which decreased the pain level. Tocilizumab is a candidate for additional treatment of juvenile idiopathic arthritis-like arthritis associated with childhood-onset MCTD.
BackgroundCalcifying fibrous tumor (CFT) arising from the pleura is a relatively rare benign lesion in young and middle-aged adults. We report a 31-year-old woman with pleural CFT who underwent successful complete thoracoscopic enucleation.Case presentationAn asymptomatic woman presented with a mass in the right lower lung field that was incidentally detected on a chest X-ray during a routine medical checkup. Chest computed tomography showed a well-defined mass in the lower mediastinum, with a maximum diameter of approximately 5.5 cm. Esophagogastroduodenoscopy showed no abnormal findings in the esophagus. An endoscopic ultrasonography (EUS) revealed a well-defined tumor with no internal blood flow. EUS-fine needle aspiration failed to establish a definitive diagnosis. Therefore, thoracoscopic tumor enucleation was performed for diagnostic and therapeutic purposes. Based on the histopathological findings of the resected specimen, the presence of a tumor with a high fibrous component in a young woman, and the identification of granulomatous calcifications, a diagnosis of CFT was established.ConclusionsComplete thoracoscopic tumor enucleation was successfully performed for CFT arising from the pleura in a young adult woman.
Smoking-related interstitial lung diseases (SRILDs) are a group of heterogeneous diffuse pulmonary parenchymal diseases associated with tobacco exposure. Smoking-related interstitial fibrosis (SRIF) is relatively recent, a pathologically defined form of SRILDs. SRIF is characterized by the accumulation of macrophages in the alveolar spaces, which is associated with interstitial inflammation and fibrosis. The macrophages frequently contain light brown pigment and are called 'smoker's macrophages'. Patients with SRIF who have clinical evidence of interstitial lung disease are most commonly relatively young, heavy smokers with abnormalities on chest computed tomography showing ground-glass opacities, peripheral consolidation, and reticulation. Although SRIF is caused by cigarette smoking, the exact pathophysiological mechanisms by which smoking causes this type of interstitial fibrosis remain unknown. The degree of fibrosis and appearance of macrophage aggregates are important points of distinction when evaluating and diagnosing SRIF. Macrophage heterogeneity, particularly the activation and function of monocyte-derived alveolar macrophages (Mo-AMs) and interstitial macrophages (IMs), has important implications for the pathogenesis of SRIF and developing treatments. Further researches focused on smoker's macrophages are needed to understand of the pathogenesis of SRIF.
Introduction We present the case of a rapidly growing inferior vena cava tumor thrombus in renal cell carcinoma. Case presentation We present a case of a 66‐year‐old woman with right renal cell carcinoma with a tumor thrombus extending 2 cm into the inferior vena cava on an initial Imaging. Radical surgery was performed 6 weeks after the first visit. Intraoperatively, the tumor thrombus was confirmed to have grown near the diaphragm. The tumor was resected using an inferior vena cava clamping just below the diaphragm. The tumor thrombus and renal cell carcinoma were completely removed. There was no recurrence 6 months postoperatively. Conclusion Inferior vena cava tumor thrombus in renal cell carcinoma can grow in a short period, suggesting that preoperative imaging evaluation at the appropriate time is important. Once inferior vena cava tumor thrombus of renal cell carcinoma occurs, surgery should not be delayed unless there is an urgent reason.
IntroductionWe encountered a case of urinary retention caused by a urethral caruncle.Case presentationAn 86‐year‐old woman presented to our hospital with urinary retention. When the urinary bladder catheter was placed, a smooth, well‐defined mass 20 mm in diameter was detected on the posterior wall of the external urethral meatus. The patient was diagnosed with urinary retention due to a urethral caruncle, and the mass was resected. The mass was pathologically compatible with a urethral caruncle. The patient could urinate postoperatively. Ultrasound test after surgery showed residual urine volume was 100 mL.ConclusionInspecting the urethral meatus is vital in the clinical examination of older women with voiding symptoms. A urethral caruncle is a rare cause of urinary retention. However, large urethral caruncles are at risk of causing urinary retention suggesting that resecting the urethral caruncles at an appropriate time is desirable.
Immune check point inhibitors (ICIs) have durable antitumor effects. However, autoimmune toxicities, termed immune-related adverse events, occur in some patients. We report a case of severe immune aplastic anemia (AA) in a patient with non -small cell lung cancer who was receiving atezolizumab with bevacizumab/carboplatin/paclitaxel. Although the cancer has not recurred, his bone marrow is depleted and he did not respond to immunosuppressive therapy. He has survived for 1.5 years with blood transfusions and infection control. Immune AA associated with ICIs is rare, and a treatment has not yet been established. This case report provides information on the management and treatment response of patients with AA caused by ICIs. Further studies should investigate the mechanism and pathogenesis of immune AA caused by ICIs.
Background and AimSome patients with functional gastrointestinal disorders exhibit pancreatic dysfunctions and pancreatic enzyme abnormalities. Thus, we aimed to clarify whether significant differences in clinical characteristics, prevalence of pancreatic enzyme abnormalities, duodenal inflammation, and protease-activated receptor 2 (PAR2) expression levels related to hypersensitivity exist between functional dyspepsia (FD) alone and FD-irritable bowel syndrome (IBS) overlap group. MethodsNinety-three patients based on the Rome IV criteria, FD alone (n = 44) and FD overlapped with IBS (n = 49) group were enrolled. The patients scored their own clinical symptoms after consuming high-fat meals. Serum trypsin, PLA2, lipase, p-amylase, and elastase-1 levels were measured. PAR2, eotaxin-3, and TRPV4 mRNA levels in duodenum were determined using real-time polymerase chain reaction methods. PRG2- and PAR2 in the duodenum were evaluated using immunostaining. ResultsFD score and global GSRS in patients with FD-IBS overlap were significantly higher than FD alone. Although the prevalence of pancreatic enzyme abnormalities in patients with FD alone was significantly (P < 0.01) higher than that in FD-IBS overlap, the ratio of aggravation of clinical symptoms following high-fat intake in patients with FD-IBS overlap was significantly higher (P = 0.007) than that in patients with FD alone. PAR2- and PRG2-double positive cells were localized in the degranulated eosinophils in the duodenum of patients with FD-IBS overlap. The number of PAR2- and PRG2-double positive cells in FD-IBS overlap was significantly (P < 0.01) higher than FD alone. ConclusionsPancreatic enzyme abnormalities and PAR2 expression on degranulated eosinophils infiltrations in the duodenum may be associated with the pathophysiology of patients with FD-IBS overlap in Asian populations.
Perineural spread (PNS) from pelvic carcinoma has been regarded as a pathway to muscle and bone metastasis. However, few cases have been reported, especially in patients with bladder carcinoma. In the present report, we discuss a case of diffuse cancer involvement in the muscle 5 years after radical cystectomy for advanced bladder carcinoma. Careful observation of temporal changes on medical images confirmed PNS as the pathway to muscle metastasis (i.e., primary PNS). Our report presents early and post-treatment CT, MRI and FDG-PET/CT findings of PNS from the bladder carcinoma.
BackgroundA neurofibroma is a benign tumor that arises from Schwann cells and neurofibromas occur throughout the skin of neurofibromatosis type 1 (NF-1: Von Recklinghausen's disease) patients. A retroperitoneal solitary neurofibroma without any clinical signs of NF1 has been rarely reported. Herein, we present a case of a retroperitoneal solitary neurofibroma mimicking lymph node metastasis of colon cancer as well as a literature review.Case presentationAn 80-year-old woman with abdominal pain and nausea was transported and diagnosed with bowel obstruction arising from sigmoid colon cancer A colonic stent was inserted to alleviate the bowel obstruction. A computed tomography scan with contrast revealed a liver tumor in segment 3, and an enlarged lymph node around the abdominal aorta. Whole-body 18F-fluorodeoxyglucose-positron emission tomography-CT (FDG-PET-CT) examine revealed increased FDG uptake in the liver tumor and enlarged lymph node. Liver and distant lymph node metastasis were diagnosed and we made a plan for a two-stage operation of the colon cancer and the metastatic lesions because laparotomy resection was needed for the retroperitoneal lymph node. Laparoscopic sigmoid colectomy was performed first. Pathological examination showed a tubular adenocarcinoma. A laparotomy for the metastatic lesions was performed to ensure complete lymph node dissection secondly. Histopathological findings of the liver tumor showed metastasis of sigmoid colon cancer. However, the tissue regarded as the enlarged lymph node was diagnosed as a neurofibroma. No metastasis and recurrence were observed.ConclusionAlthough most neurofibromas are benign, malignant transformation of a neurofibroma is possible. PET-CT showed our patient had a high accumulated retroperitoneal tumor co-existing with colon cancer and liver metastasis. The treatment strategy of a solitary neurofibroma must be selected carefully considering the site of occurrence and the patient's background and aggressive resection of a tumor co-existing with another malignant tumor is needed.
INTRODUCTION:Desmoplastic malignant pleural mesothelioma (DMPM) is a sarcoma-type mesothelioma, comprising approximately 5% of malignant pleural mesotheliomas. Although effusion cytology is commonly used as the primary diagnostic approach for mesothelioma, it may not be useful for DMPM because of the presence of desmoplasia and bland cellular atypia. We report a case, and previously undescribed cytological features, of DMPM that was diagnosed during autopsy.CASE PRESENTATION:A man in his 60s with a history of occupational asbestos exposure was referred to our hospital with right chest pain. A chest CT scan showed right pleural effusion. Thirteen months later, the patient died of respiratory failure. During autopsy, scrape-imprint smears were prepared and cytology of pleural effusions was performed. The scrape-imprint smear samples showed spindle cells with mild nuclear atypia and grooves with fibrous stroma. Pleural effusion cytology revealed spindle cells with mild nuclear atypia, as well as grooves with loose epithelial connections. Histological examination of the right pleura showed spindle cells proliferating with dense collagen fibers, as seen in the cytological samples, thus indicating a diagnosis of DMPM, which was confirmed by fluorescence in situ hybridization.CONCLUSION:Cytological procedures such as pleural effusion cytology and scrape-imprinting cytology may help in diagnosing rare tumors such as DMPM.
The incidence of papillary thyroid carcinoma (PTC) is increasing worldwide. The biomarkers to identify aggressive types of PTC are limited, illustrating the need to establish reliable novel biomarkers. Protein disulfide isomerase A3 (PDIA3) is a chaperone protein that modulates the folding of newly synthesized glycoproteins and stress-responsive proteins in the endoplasmic reticulum. Although the role of PDIA3 in various cancers such as breast, uterine cervix, head and neck, and gastrointestinal tract has been examined, its expression in thyroid cancer has not been reported. We retrospectively reviewed accumulated data with long-term follow-up of 1,139 PTC patients, and investigated the correlation between immunohistochemical expression of PDIA3 in PTC patients and clinicopathological features and prognosis. PDIA3 expression was significantly lower in PTCs compared to normal thyroid tissues (NTT; n = 80, p = 0.002). In PTCs, correlation between low PDIA3 expression and lymph node metastasis (p = 0.018) and the number of positive nodes (p = 0.004) was observed. Patients with low PDIA3 expression exhibited worse cause-specific survival compared to those with high PDIA3 expression (p = 0.013). Our findings indicate that low PDIA3 expression is related to poor clinical outcome in PTC patients, and that PDIA3 may potentially be a novel ancillary biomarker. Further clarification of the biological role of PDIA3 in PTC is warranted for the future clinical application.
We have reported that refractory functional dyspepsia patients with pancreatic enzyme abnormalities (FD-P). We tried to analyze the prevalence of exocrine pancreatic insufficiency (EPI) in FD-P patients to clarify whether the pathophysiology of FD patients including clinical symptoms and quality of life were associated with EPI. We enrolled forty-nine patients presenting with typical symptoms of FD-P patients (n = 20) and asymptomatic patients with pancreatic enzyme abnormalities (AP-P) (n = 29). Five pancreatic enzymes (p-amylase, lipase, elastase-1, trypsin, and PLA2) were measured and STAI-state/-trait and SF-8 were evaluated. Pancreatic exocrine function was analyzed using N-benzoyl-l-tyrosyl-p-aminobenzoic acid (BT-PABA). There were no significant differences in patient background between FD-P and AP-P patients. BT-PABA test scores for FD-P patients (61.67 ± 5.55) were significantly (p = 0.01) lower than in AP-P patients (95.38 ± 2.36). Physical component scale (PCS) in FD-P patients was significantly (p = 0.002) lower than that in AP-P patients. STAI-state was relatively (p = 0.054) associated with BT-PABA test in FD-P and AP-P patients by multiple logistic regression analysis. The prevalence of EPI in FD-P patients was significantly higher than that in AP-P patients and was relatively associated with state of anxiety. Further studies will be needed to clarify how EPI or pancreatic enzyme abnormalities are associated with the pathophysiology of FD-P patients.