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Context: Paragangliomas located within the pericardium represent a rare yet challenging clinical situation. Objective: The current analysis aimed to describe the clinical characteristics of cardiac paragangliomas, with emphasis on the diagnostic approach, genetic background, and multidisciplinary management. Methods: Twenty-four patients diagnosed with cardiac paraganglioma (PGL) in Peking Union Medical College Hospital, Beijing, China, between 2003 and 2021 were identified. Clinical data was collected from medical record. Genetic screening and succinate dehydrogenase subunit B immunohistochemistry were performed in 22 patients. Results: The median age at diagnosis was 38 years (range 11-51 years), 8 patients (33%) were females, and 4 (17%) had familial history. Hypertension and/or symptoms related to catecholamine secretion were present in 22 (92%) patients. Excess levels of catecholamines and/or metanephrines were detected in 22 (96%) of the 23 patients who have completed biochemical testing. Cardiac PGLs were localized with 131I-metaiodobenzylguanidine scintigraphy in 11/22 (50%), and 99mTc-hydrazinonicotinyl-tyr3-octreotide scintigraphy in 24/24 (100%) patients. Genetic testing identified germline SDHx mutations in 13/22 (59%) patients, while immunohistochemistry revealed succinate dehydrogenase (SDH) deficiency in tumors from 17/22 (77%) patients. All patients were managed by a multidisciplinary team through medical preparation, surgery, and follow-up. Twenty-three patients received surgical treatment and perioperative death occurred in 2 cases. Overall, 21 patients were alive at follow-up (median 7.0 years, range 0.6-18 years). Local recurrence or metastasis developed in 3 patients, all of whom had SDH-deficient tumors. Conclusion: Cardiac PGLs can be diagnosed based on clinical manifestations, biochemical tests, and appropriate imaging studies. Genetic screening, multidisciplinary approach, and long-term follow-up are crucial in the management of this disease.
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Thrombus formation on a well-conserved bicuspid aortic valve is rare. We encountered a patient with organized thrombus formation on a native bicuspid aortic valve without calcification or stenosis, which was found occasionally during an elective operation for ascending aorta replacement surgery. The location of the thrombus was just at the orifice of left coronary artery, which produced the atherosclerosis-like symptoms such like exertional chest tightness and dyspnea. And these are no apparent predisposing causes of thrombosis could be ascertained postoperatively. The patient is in excellent condition 6 months after the operation. The lesson we learned from our case is that when the patient's symptom can't correspond with his or her diagnosis, we should ask more questions, evaluate the patient thoroughly and make the differential diagnosis as possible as we can. And the surgery can be performed aggressively when patient's symptoms cannot be figured out by physical examination, not only for pathologic confirmation but also for the prevention of life-threatening complications that can caused by either condition.
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Background: Cardiac sarcomas are rare malignancies with a poor prognosis. Although angiosarcoma is the most common histological subtype, its features are poorly characterized. This study aimed to compare the clinical characteristics of the various cardiac sarcomas and the surgical techniques used and to identify factors influencing the prognosis. Methods: Forty patients who underwent surgery for cardiac sarcomas were included; 60% of them had angiosarcoma. Clinical characteristics, tumor location, surgical techniques used, and the prognosis were compared between patients with angiosarcoma and patients with other subtypes. Kaplan-Meier curves and multivariable Cox regression were used to identify predictors of postoperative survival. Results: Angiosarcomas were more likely than the other subtypes to present as pericardial effusion (85% vs 50%, P = .014). Early surgery was performed (median 24.0 days) regardless of histological subtype. The surgical technique varied according to histological subtype. Mean postoperative survival was 10 months. A positive margin (P = .13), high Ki-67 index (P = .19), younger age (P = .86), and angiosarcoma (P = .87) were identified to be potentially poor prognostic factors in univariate analyses. Cox regression identified R0 resection to be the only significant independent predictor of the prognosis after surgery (hazard ratio, 0.423, P = .039). Conclusions: Angiosarcoma differs from other subtypes of cardiac sarcoma in terms of clinical symptoms, tumor location, surgical techniques used, and prognosis. Early surgery is needed regardless of subtype. R0 resection is the only independent predictor of postoperative survival, and complete resection is usually achievable. The prognosis may be poorer in patients with a positive margin, high Ki-67 index, younger age, and angiosarcoma.
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Objectives: This retrospective study aimed to summarize the feasibility and experience of utilizing a one-stage operation via single laparotomy to treat intracardiac leiomyomatosis (ICL). Materials and methods: A retrospective study of 13 patients with ICL who underwent one-stage resections was conducted at Peking Union Medical College Hospital from June 2015 to December 2021. All patients had their tumors removed by single laparotomy and were divided into a short venotomy group (6 cases) and an extensive venotomy group (7 cases). We reviewed the patient characteristics, surgical procedures, postoperative pathology, and perioperative and follow-up outcomes of all patients. Results: All patients underwent surgery for ICL resection using single laparotomy with a 100% success rate. Two patients had tumors distal to the right ventricle (RV), 2 patients had tumors that protruded into the RV in diastole and were confined to the right atrium (RA) in systole, and the other 9 patients had tumors confined to the RA that did not involve the tricuspid valve. The tumor was completely resected in 10 patients, yet 3 patients had a residual tumor. Six patients completed the surgery with short venotomy, 7 completed the surgery with extensive venotomy, and 9 underwent simultaneous total hysterectomy and bilateral adnexal resection. The mean operative time was 370.8 ± 111.0 min, and the mean blood loss was 992.3 ± 994.5 mL. Intraoperative blood loss was lower (483.3 ± 213.7 ml vs. 1429.2 ± 1208.0 ml; P = 0.020) and operative time was shorter (286.5 ± 71.9 min vs. 443.1 ± 84.4 min; P=0.004) in the short venotomy group than in the extensive venotomy group. At a mean follow-up of 26.3 ± 18.8 months, 1 patient had a local recurrence in the pelvis, and 1 patient died of pancreatic cancer, while the remaining patients had no recurrence during follow-up. Conclusion: One-stage resection of ICL patients by means of a single laparotomy is feasible and effective.
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OBJECTIVES: To explore the personalized treatment strategy of sternal fixation and closure of sternal median incision in open cardiac surgery. METHODS: A total of 293 patients who underwent open-heart surgery with a median sternal incision at Peking Union Medical College Hospital from January 2019 to March 2021 were divided into two groups, according to the timing and type of treatment. The first 169 patients received single-wire fixation and closure (control group), while the subsequent 124 patients received double-wire fixation and closure (study group). The patients were followed up for three months to observe the duration of pain, sternal instability, and occurrence of chest wound infection. RESULTS: The average age was 53±30 years in the control group and 55±34 years in the study group (P = 0.594). There were no significant differences in baseline data between the two groups (P > 0.05). Compared with the control group, the study group had a shorter duration of pain (P < 0.05), smaller drainage volume within three days postoperatively (650 ml vs. 770 ml, P < 0.05), lower incidence of superficial sternal wound infection (2.4% vs. 8.9%, P = 0.042), and lower incidence of sternal instability (1.6% vs. 8.3%, P = 0.026). Deep sternal wound infection occurred in two patients in the control group and none in the study group; however, this difference was not significant. No surgery-related deaths occurred. CONCLUSIONS: Selecting the appropriate sternal fixation and closure method, according to the characteristics of patients, can reduce the incidence of sternal incision complications. We proposed a personalized selection strategy for sternal fixation and closure, which requires verification in clinical studies.
Subject(s)
Cardiac Surgical Procedures , Surgical Wound Infection , Adult , Aged , Aged, 80 and over , Cardiac Surgical Procedures/methods , Humans , Middle Aged , Pain/complications , Sternotomy/adverse effects , Sternotomy/methods , Sternum/surgery , Surgical Wound Infection/epidemiology , Treatment Outcome , Young AdultABSTRACT
A 47-year-old previously healthy man was referred to a local hospital with chest tightness, oliguria, and lower extremity edema for seven days. An initial investigation revealed acute heart failure and kidney injury. The patient was intensively treated with cardiac and renal replacement therapy, and cardiorenal function improved one week later. Two months later, echocardiography was performed because chest tightness and edema had not resolved. Echocardiography showed Valsalva sinus rupture, and the patient was transferred to our center. Myocardial calcification was observed in the left ventricular wall on computed tomography after admission. The patient underwent cardiac surgery and recovered smoothly. At the three-year follow up, the patient was asymptomatic with normal renal function and serum electrolytes. Imaging revealed a significant reduction in diffuse calcification of the left ventricular wall. This case indicates that this rare form of reversible myocardial calcification may be associated with acute heart and renal failure caused by Valsalva sinus rupture. The results of this case will guide clinicians about further management and timely referral of such patients to appropriate specialties.
Subject(s)
Aortic Aneurysm , Aortic Rupture , Heart Failure , Sinus of Valsalva , Aortic Aneurysm/surgery , Aortic Rupture/complications , Aortic Rupture/diagnosis , Heart Failure/complications , Heart Failure/etiology , Humans , Kidney/physiology , Male , Middle Aged , Sinus of Valsalva/diagnostic imaging , Sinus of Valsalva/surgeryABSTRACT
BACKGROUND AND AIM: To determine the factors contributing to successful mitral valve repair (MVP) and to discuss the effect of complex techniques on the durability of MVP for active infective endocarditis (IE) affecting the mitral valve. METHODS: One hundred and eighty-seven patients were enrolled; 39.6% underwent mitral valve replacement (MVR) and 60.4% underwent MVP. We used logistic regression to identify influencing factors of the choice of surgical technique. The results were compared between groups and subgroups after propensity score matching (PSM). RESULTS: Risk factors for MVR included poor valve quality (odds ratio [OR] 23.3, p = .001), a large defect after debridement (OR 16.4, p < .001), and heavy valve infection (OR 3.7, p = .027). After PSM, we did not find a significant difference in the frequency of major postoperative complications or the in-hospital or postdischarge death rate. The reintervention rate for MVP was significantly higher than that for MVR (p = .047). Subgroup analysis found a significant relationship between the use of a complex repair technique and the need for reoperation (p = .020). CONCLUSIONS: The choice of valve repair or replacement for patients with active IE affecting the mitral valve was influenced by the intraoperative characteristics of the infected valve rather than the severity of systemic infection or overall health status. The choice of surgical treatment strategy had no effect on major postoperative complications, in-hospital mortality, or medium-term survival. However, the medium-term durability of MVP was poorer than that of MVR. The use of the patch technique for free margins or extensive leaflet defects was associated with a need for reintervention.
Subject(s)
Endocarditis, Bacterial , Endocarditis , Heart Valve Prosthesis Implantation , Mitral Valve Insufficiency , Aftercare , Endocarditis/etiology , Endocarditis/surgery , Endocarditis, Bacterial/surgery , Heart Valve Prosthesis Implantation/methods , Humans , Mitral Valve/surgery , Mitral Valve Insufficiency/surgery , Patient Discharge , Postoperative Complications/epidemiology , Postoperative Complications/etiology , Postoperative Complications/surgery , Treatment OutcomeABSTRACT
BACKGROUND: Infective endocarditis (IE)-associated rapidly progressive glomerulonephritis (RPGN) is rarely reported. Sporadic case reports have noted the diagnostic and therapeutic challenge in IE-associated glomerulonephritis because it may masquerade as idiopathic vasculitis. METHODS: Patients with clinical diagnosis of IE-related RPGN in a tertiary hospital in China between January 2004 and May 2021 were identified and retrospectively reviewed. RESULTS: Twenty-four patients with IE-associated RPGN were identified. All patients presented with fever and multiorgan system involvement on top of heart and kidneys, spleen (79%, 19/24), skin (63%, 15/24), lung (33%, 8/24) and nervous system (17%, 4/24). Six of the 24 patients (25%) were initially suspected to have ANCA-associated or IgA vasculitis. Forty-five percent of patients are seropositive for ANCA. Renal histology showed mesangial and/or endocapillary hypercellularity with extensive crescents in most patients. C3-dominant deposition was the predominant pattern on immunofluorescence and pauci-immune necrotising crescentic glomerulonephritis was observed in one case. All patients received antibiotics with or without surgery. Six patients received immunosuppressive therapy before antibiotics due to misdiagnosis and seven patients received immunosuppressive therapy after antibiotics due to persistence of renal failure. Three of the 24 patients died due to severe infection. All the surviving patients had partial or complete recovery of renal function. CONCLUSION: IE-associated RPGN is rare and the differential diagnosis from idiopathic vasculitis can be challenging due to overlaps in clinical manifestations, ANCA positivity and absence of typical presentations of IE. The prognosis is generally good if antibiotics and surgery are not delayed. The decision on introducing immunoruppressive treatment should be made carefully on a case by case basis when kidney function does not improve appropriately after proper anti-infective therapy.Key messagesInfective endocarditis associated RPGN is rare and differentiating it from idiopathic vasculitis can be challenging due to overlap in clinical manifestations, ANCA positivity and occasional absence of typical manifestations of infective endocarditis.Kidney function usually responds to antibiotic therapy alone.Immunosuppressive therapy may be beneficial in carefully selected patients whose kidney function does not improve with antibiotics alone.
Subject(s)
Endocarditis , Glomerulonephritis , Vasculitis , Endocarditis/complications , Glomerulonephritis/diagnosis , Glomerulonephritis/drug therapy , Humans , Kidney/pathology , Retrospective Studies , Vasculitis/complications , Vasculitis/diagnosis , Vasculitis/drug therapyABSTRACT
BACKGROUND: The coronavirus disease 2019 (COVID-19) pandemic has been and will continue to be a challenge to the healthcare system worldwide. In this context, we aimed to discuss the impact of the COVID-19 pandemic on the diagnosis, timing, and prognosis of surgical treatment for active infective endocarditis (IE) during the pandemic and share our coping strategy. METHODS: A total of 39 patients were admitted for active IE in the year 2020. The number of the same period last year was 50. Medical information of these two groups was extracted from our surgical database. Data were compared between the two groups and differences with or without statistical significance were discussed. RESULTS: In the pandemic year, we admitted fewer transferred patients (64.1% vs. 80%, p = .094). Timespan for diagnosis were prolonged (60 vs. 34.5 days, p = .081). More patients were admitted in emergency (41% vs. 20%, p = .030) More patients had heart failure (74.4% vs. 40%, p = .001), sepsis (69.2% vs. 42.0%, p = .018), or cardiogenic shock (25.6% vs. 8.0%, p = .038). Overall surgical risk (EuroSCORE II) was higher (4.15% vs. 3.24%, p = .019) and more commando surgery was performed (7.7% vs. 2.0%, p = .441). However, we did not see more postoperative complications, and early mortality was not worse either (0 vs. 4%, p = .502). CONCLUSIONS: The negative impact of the COVID-19 pandemic on the clinical practice of surgical treatment for active IE was multifaceted. However, with the preservation of the effectiveness of multidisciplinary IE surgical team, the early outcomes were comparable with those in the normal years.
Subject(s)
COVID-19 , Endocarditis, Bacterial , Endocarditis , Endocarditis/surgery , Endocarditis, Bacterial/surgery , Humans , Pandemics , Postoperative Complications/epidemiology , Retrospective Studies , Treatment OutcomeABSTRACT
Cardiac tumors are rare. They were found in only 0.001%-0.300% of cases in a relatively recently reported autopsy series. Among cardiac tumors, primary hemangioma accounted for approximately 2.8% of all primary resected tumors, indicating this is a particularly rare benign neoplasm. We present a patient with a 5×3×2 cm cavernous hemangioma, arising from the right atrial roof and occupying the atrial septum and inseparable from the aortic root. We successfully accomplished a complete surgical resection of a cardiac cavernous hemangioma and reconstructed the cardiac atrium by a bovine pericardial patch.
Subject(s)
Atrial Septum , Heart Neoplasms , Hemangioma, Cavernous , Hemangioma , Humans , Animals , Cattle , Hemangioma, Cavernous/diagnosis , Hemangioma, Cavernous/surgery , Hemangioma, Cavernous/pathology , Hemangioma/pathology , Hemangioma/surgery , Heart Atria/surgery , Heart Atria/pathology , Heart Neoplasms/diagnosis , Heart Neoplasms/surgery , Heart Neoplasms/pathologyABSTRACT
OBJECTIVE: In the present study, we analyzed the advantages and feasibility of non-open-heart surgery without cardiopulmonary bypass for intracardiac intravenous leiomyomatosis. METHODS: We retrospectively reviewed 23 cases of intracardiac intravenous leiomyomatosis and divided them into a noncardiopulmonary bypass (NCPB) group (9 cases) and a cardiopulmonary bypass (CPB) group (14 cases) according to the surgical treatment received. The clinical characteristics and anatomic features, including the diameter of the tumor, right atrium, and inferior vena cava, were recorded, and the perioperative data, including the operation time, blood loss, postoperative hemoglobin change, and follow-up results, were analyzed and compared between the two groups. RESULTS: The NCPB group had required a shorter operation time (321.9 ± 104.2 minutes vs 526.3 ± 95.6 minutes; P < .001) and had experienced less blood loss (456.3 ± 249.9 mL vs 815.4 ± 435.6 mL; P = .048) compared with the CPB group. The NCPB group had a small maximum cross-sectional area of the tumor inside the right atrium (475.5 ± 509.6 mm2), a low proportion of the maximum cross-sectional area of the entrance of the right atrium (average, 26.1%), no tricuspid valve or atrial wall involvement, and high mobility inside the inferior vena cava and heart chamber. All 23 patients had recovered well postoperatively, and no recurrence had developed during 24 months of follow-up. CONCLUSIONS: For intravenous leiomyomatosis with a smaller cross-sectional area in the right atrium that can be mobilized, surgery without CBP is feasible and should be considered.
Subject(s)
Cardiac Surgical Procedures , Cardiopulmonary Bypass , Heart Atria/surgery , Leiomyoma/surgery , Uterine Neoplasms/surgery , Vena Cava, Inferior/surgery , Adult , Cardiac Surgical Procedures/adverse effects , Cardiopulmonary Bypass/adverse effects , Feasibility Studies , Female , Heart Atria/diagnostic imaging , Heart Atria/pathology , Humans , Leiomyoma/diagnostic imaging , Leiomyoma/pathology , Middle Aged , Operative Time , Postoperative Complications/etiology , Retrospective Studies , Time Factors , Treatment Outcome , Uterine Neoplasms/diagnostic imaging , Uterine Neoplasms/pathology , Vena Cava, Inferior/diagnostic imaging , Vena Cava, Inferior/pathologyABSTRACT
BACKGROUND: Purpura and glomerulonephritis are typical presentations in IgA vasculitis. Infective endocarditis mimicking IgA vasculitis by presenting with glomerulonephritis and purpura is rarely reported. METHODS: We searched for cases with infective endocarditis-associated purpura and glomerulonephritis in a tertiary hospital in China and retrospectively reviewed their clinicopathological features. Differential diagnosis and treatment in patients with infective endocarditis-associated purpura and glomerulonephritis were discussed. RESULTS: A total of 20 cases with infective endocarditis-associated purpura and glomerulonephritis were identified among 548 cases with infective endocarditis in our center during an 8-year period: 7 of the 20 cases (35%) were initially misdiagnosed as IgA vasculitis and 10 cases (50%) presented with left-sided endocarditis caused by Streptococcus viridans. Fever (100%, 20 out of 20), prior valvular deformities (80%, 16 out of 20), cardiac murmur (95%, 19 out of 20), splenomegaly (84%, 16 out of 19), embolism (55%, 11 out of 20), and hypocomplementemia (76%, 13 out of 17) were present in most patients. Crescents and mesangial hypercellularity with or without endothelial hypercellularity were the primary findings on light microscopy, with C3-dominant deposition on immunofluorescence. But IgA-dominant staining was also observed (40%, 2 out of 5). In patients with rapidly progressive glomerulonephritis, patients with complete recovery of renal function had shorter disease duration and higher ratio (67% vs 20%) of immunosuppressive therapy compared with patients with partial recovery. CONCLUSIONS: Infective endocarditis-associated glomerulonephritis and purpura can closely mimic IgA vasculitis. Differential diagnosis is challenging, particularly when typical presentations of infective endocarditis are absent. In adults with presentations like IgA vasculitis, infective endocarditis should be evaluated through comprehensive clinical and pathological investigations. Immunosuppressive therapy can be considered in patients with severe glomerulonephritis who do not improve after proper anti-infective therapy.
Subject(s)
Endocarditis/diagnosis , Glomerulonephritis/physiopathology , IgA Vasculitis/diagnosis , Purpura/physiopathology , Streptococcal Infections/diagnosis , Adult , Antibodies, Antineutrophil Cytoplasmic/blood , Antibodies, Antinuclear/blood , Antibodies, Antiphospholipid/blood , Autoantibodies/blood , Complement System Proteins/metabolism , Diagnosis, Differential , Endocarditis/blood , Endocarditis/complications , Endocarditis/physiopathology , Female , Glomerulonephritis/blood , Glomerulonephritis/etiology , Glomerulonephritis/pathology , Humans , Immunosuppressive Agents/therapeutic use , Male , Middle Aged , Purpura/blood , Purpura/etiology , Rheumatoid Factor/blood , Streptococcal Infections/blood , Streptococcal Infections/complications , Streptococcal Infections/physiopathology , Thrombocytopenia/blood , Viridans Streptococci , Young AdultABSTRACT
INTRODUCTION: Intravenous leiomyomatosis (IVL) is currently regarded as a special variant of the common uterine leiomyoma (LM). Though IVL shows a similar histological morphology to LM, IVL is characterized by unique intravenous growth patterns and low-grade malignant potential, which are quite different from LM. There are currently few studies underlying the molecular alterations of IVL, though this information is important for understanding the pathogenesis of the disease, and for identifying potential biomarkers. METHOD: We carried out a high-throughput whole transcriptome sequencing of tumor and normal tissue samples from five IVL patients and five LM patients and compared the differentially expressed genes (DEGs) between IVL and leiomyoma. We performed multiple different enrichment and target analyses, and the expression of selected DEGs was validated using RT-qPCR in formalin-fixed samples. RESULTS: Our study identified substantial different genes and pathways between IVL and LM, and functional enrichment analyses found several important pathways, such as angiogenesis and antiapoptosis pathways, as well as important related genes, including SH2D2A, VASH2, ADAM8, GATA2, TNF, and the lncRNA GATA6-AS1, as being significantly different between IVL and LM (P = .0024, P = .0195, P = .0212, P = .0435, P = .0401, and P = .0246, respectively). CXCL8, LIF, CDKN2A, BCL2A1, COL2A1, IGF1, and HMGA2 were also differently expressed between IVL and LM groups, but showed no statistical difference (P = .2409, P = .1773, P = .0596, P = .2737, P = .1553, P = .1045, and P = .1847, respectively) due to the large differences among individuals. Furthermore, RT-qPCR results for five selected DEGs in IVL tissues and adjacent nontumor tissues were mainly consistent with our sequencing results. CONCLUSION: Our results indicated that IVL may be a solid entity that is unique and different from LM, proving consistent with previous studies. Furthermore, we identified DEGs, particularly within angiogenesis and antiapoptosis pathway-related genes that may play crucial roles in the development and pathogenesis of IVL and may be potential specific biomarkers.
Subject(s)
Leiomyomatosis/genetics , RNA-Seq/methods , Reverse Transcriptase Polymerase Chain Reaction , Uterine Neoplasms/genetics , Vascular Neoplasms/genetics , Apoptosis/genetics , Case-Control Studies , Female , Humans , Immunohistochemistry , Leiomyomatosis/blood supply , Leiomyomatosis/diagnostic imaging , Leiomyomatosis/pathology , Middle Aged , Neovascularization, Pathologic/genetics , Uterine Neoplasms/blood supply , Uterine Neoplasms/diagnostic imaging , Uterine Neoplasms/pathology , Vascular Neoplasms/blood supply , Vascular Neoplasms/diagnostic imaging , Vascular Neoplasms/pathology , Exome SequencingABSTRACT
Intravenous leiomyomatosis (IVL) with intracardiac involvement is a rare condition. Previous case reports have described direct tumor extension into the vena cava and cardiac chamber. Six patients with intracardiac lesion without direct growth from the IVC were admitted. Surgical resections were performed including total hysterectomy and bilateral salpingo-oophorectomy. Post-operative pathology revealed intravenous leiomyomatosis. The endothelial neoplastic implantation of IVL is a possible reason for the disseminated lesions of IVL within venous system. Awareness of this entity helps clinicians avoid misdiagnosis and take appropriate treatment.
Subject(s)
Endothelial Cells/pathology , Leiomyomatosis/pathology , Myocardium/pathology , Uterine Neoplasms/pathology , Vena Cava, Inferior/pathology , Adult , Cardiac Surgical Procedures , Female , Humans , Hysterectomy , Leiomyomatosis/surgery , Neoplasm Invasiveness , Salpingo-oophorectomy , Treatment Outcome , Uterine Neoplasms/surgery , Vascular Surgical Procedures , Vena Cava, Inferior/surgeryABSTRACT
BACKGROUND: Food-borne trematodiases are an important group of neglected global diseases. Affected patients in regions with low prevalence usually experience delayed diagnosis, especially when presenting with atypical clinical symptoms. Here, we presented a rare case of a Chinese patient infected with three food-borne trematodiases. CASE PRESENTATION: A 42-year-old man presented with diarrhea, lower extremity edema, and symptoms of cardiac dysfunction. He had a history of intermittent consumption of raw freshwater fishes for 6-7 years. Upon evaluation, he had eosinophilia, anemia, intrahepatic bile duct dilatation and a growing space-occupying lesion in the left atrium. The patient underwent a cardiac surgery which revealed an endocardial hematoma due to mechanical injuries. Imaging investigations also revealed intracranial and pulmonary lesions. A total of three trematodiases were diagnosed based upon microscopic stool examination, from which eggs of Clonorchis sinensis, Heterophyidae and Echinostomatidae were identified. Deposition of Clonorchis sinensis eggs was also observed from ileocecal squash slides. The patient was successfully treated with three cycles of praziquantel. CONCLUSIONS: Food-borne trematodiases may present with systemic involvement. Patients with dietary history of high risk or atypical ingestions should be evaluated for parasitic infection, even in non-endemic areas.
Subject(s)
Food Parasitology , Foodborne Diseases/parasitology , Neglected Diseases/parasitology , Trematode Infections/parasitology , Adult , Animals , Anthelmintics/therapeutic use , Fishes/parasitology , Foodborne Diseases/drug therapy , Foodborne Diseases/pathology , Humans , Male , Neglected Diseases/drug therapy , Neglected Diseases/pathology , Praziquantel/therapeutic use , Trematoda , Trematode Infections/drug therapy , Trematode Infections/pathologyABSTRACT
The purpose of this study was to explore the potential relationship between intravenous leiomyomatosis (IVL) and uterine myoma (UM) at the molecular level. RNA-sequencing was performed on IVL tumours, UM tumours, and adjacent normal uterine muscle. We compared the gene expression levels between IVL and normal uterine muscle, UM and normal uterine muscle, to identify differentially expressed genes (DEGs). Then we used Gene Ontology Enrichment Analysis to determine the functions of the DEGs and performed specimen cluster analysis. We obtained 98 DEGs between IVL and adjacent normal uterine muscle, and 61 DEGs between UM and adjacent normal uterine muscle. Functional enrichment of both IVL and UM DEGs showed that they are associated with hormone stimulus, extracellular matrix, and cell adhesion. Unsupervised clustering analysis showed that IVL and UM could not be separated completely. Among these dysregulated genes, we found that HOXA13 showed a distinct dysregulated status between IVL and UM. HOXA13 may therefore serves as a biomarker to distinguish IVL and UM. Our results showed that IVL and UM may have similar dysregulated gene networks. They may be closely related, and HOXA13 may serves as a biomarker to distinguish between IVL and UM.
Subject(s)
Leiomyomatosis/genetics , Myoma/genetics , Transcriptome , Uterine Neoplasms/genetics , Biomarkers, Tumor/genetics , Biomarkers, Tumor/metabolism , Female , Gene Regulatory Networks , Homeodomain Proteins/genetics , Homeodomain Proteins/metabolism , Humans , Leiomyomatosis/metabolism , Middle Aged , Myoma/metabolism , Uterine Neoplasms/metabolism , Uterus/metabolism , Uterus/pathologyABSTRACT
OBJECTIVES: There are few published studies on the rare disorder of intravenous-cardiac leiomyomatosis (IVCL). This study aimed to propose an individualized strategy for surgical treatment of IVCL. METHODS: In this retrospective study, we reviewed 50 patients who had undergone IVCL removal from November 2002 to October 2017 in our hospital. IVCL was classified as Type A-E according to the extent and size, with Type E being the most severe. Clinical manifestations, surgical features and follow-up data were analysed. RESULTS: Of the 50 patients in this series, 8 had Type A IVCL, 8 Type B, 29 Type C, 2 Type D and 3 Type E IVCL. One-stage removal of IVCL was performed via laparotomy without cardiopulmonary bypass (CPB) in the 8 patients with Type A, 1-stage tumour resection via sternolaparotomy under deep hypothermic arrest in 7 of the 8 patients with Type B and IVCL removal via sternolaparotomy under CPB, with 27 also under deep hypothermic arrest, in all 29 patients with Type C. Sixteen of the patients with Type C IVCL underwent staged procedures, 13 a 1-stage procedure and 21 required hepatic mobilization. All patients with Type C or E cases underwent 1-stage tumour removal via sternolaparotomy under deep hypothermic arrest. All 50 patients survived surgery. IVCL was confirmed postoperatively by histology. Ten patients had residual tumours; 9 of which did not progress. No deaths occurred during 47.8 ± 38.4 (range 1-177) months of follow-up. CONCLUSIONS: The only known curative treatment for IVCL is surgery. Herein, we present an individualized strategy for selecting surgical treatment.
Subject(s)
Cardiopulmonary Bypass/methods , Heart Neoplasms/surgery , Leiomyomatosis/surgery , Vascular Neoplasms/surgery , Adult , Aged , Cardiac Surgical Procedures , Female , Heart Neoplasms/diagnostic imaging , Heart Neoplasms/secondary , Humans , Leiomyomatosis/diagnostic imaging , Middle Aged , Ovarian Neoplasms/pathology , Ovary/blood supply , Ovary/diagnostic imaging , Retrospective Studies , Treatment Outcome , Vascular Neoplasms/diagnostic imaging , Vascular Neoplasms/pathology , Young AdultABSTRACT
Osteoporosis is a major risk factor for deep sternal wound infection, which is a rare but serious complication after median sternotomy. We investigated the incidence of deep sternal wound infection and the protective effect of bone allografts in osteoporotic patients after sternal approximation. Data were collected retrospectively from consecutive osteoporotic patients who underwent cardiac surgery via median sternotomy. Sternal approximation in the historical control group was performed with conventional steel wire sutures. Subsequent patients underwent conventional wire suturing plus bone allografting to reinforce the sternum. Perioperative management was standardized between groups. Demographics, risk factors, and postoperative outcomes were analyzed. Between January 2010 and March 2017, 284 patients underwent sternal approximation after sternotomy at our hospital. Sternal closure was completed with conventional wire sutures in the first 148 patients (Group A) and with conventional wire sutures plus bone allografting in the subsequent 136 patients (Group B). Baseline characteristics were comparable, with no significant differences between groups. Bone allografting was associated with less postoperative drainage and shorter duration of chest pain. The incidence of deep sternal wound infection was significantly lower in Group B than Group A (0.7 vs. 4.7%, P = 0.042), as was the incidence of sternal instability (0.7 vs. 7.4%, P = 0.043). Bone allografting was a reliable adjuvant method for sternal closure, associated with reduced risk of deep sternal wound infection among osteoporotic patients. Its benefits should be confirmed in larger studies.
Subject(s)
Bone Transplantation , Cardiac Surgical Procedures , Sternotomy , Sternum/surgery , Aged , Bone Transplantation/methods , Cardiac Surgical Procedures/methods , Female , Freeze Drying , Humans , Male , Middle Aged , Osteoporosis/surgery , Sternotomy/methods , Transplantation, Homologous/methods , Treatment OutcomeABSTRACT
RATIONALE: Hysterosalpingography (HSG), a standard procedure for the evaluation of women with infertility and repetitive pregnancy loss, is associated with complications such as uterine perforation, infection, allergic reactions, syncope, hemorrhage and shock, and pulmonary or retinal embolus. However, hyperthyroidism has not been reported as one of its complications. PATIENT CONCERNS AND DIAGNOSES: We report the case of a 33-year-old euthyroid woman who presented to our hospital with palpitation, hand tremor, fatigue, and excessive sweating after HSG. Thyroid function tests revealed a thyroid stimulating hormone (TSH) level of 0.012âµIU/mL (range 0.38-4.34âµIU/mL), free T4 of 2.886âng/dL (range 0.81-1.89âng/dL), and free T3 levels of 9.4âpg/mL (range 1.80-4.10âpg/mL), and antithyroglobulin antibody of 31.78âIU/mL (range <115âIU/mL). The triiodothyronine uptake was 3.057âng/mL (range 0.66-1.92âng/mL). Serum iodine (SI) and urinary iodine (UI) levels: SI of 4717.748âµg/L (range 45-90âµg/L) and UI of 18069.336âµg/L (range 26-705âµg/L). INTERVENTIONS AND OUTCOMES: The patient was diagnosed with iodine-induced hyperthyroidism (IIH), but was not treated with antithyroid drugs. She has spontaneously recovered and is pregnant currently. LESSONS: This is the first reported case of overt IIH caused by HSG in a euthyroid patient without risk factors. It suggests that HSG also leads to excessive iodine absorption, which induces secondary hyperthyroidism.