BACKGROUND Thin-walled lung cancer manifests as a cystic lesion,mostly adenocarcinoma.It is often misdiagnosed as a benign lesion in clinical practice,thus delaying the diagnosis and surgical treatment.Its natural cou...BACKGROUND Thin-walled lung cancer manifests as a cystic lesion,mostly adenocarcinoma.It is often misdiagnosed as a benign lesion in clinical practice,thus delaying the diagnosis and surgical treatment.Its natural course is rarely recorded and observed;thus,the pathogenesis and diagnosis need to be clarified and improved.CASE SUMMARY A 66-year-old man developed a mass in the upper lobe of the right lung and a small,thin-walled cavity in the lower lobe of the right lung in 2007.The right upper lobe mass was confirmed to be adenocarcinoma after surgery.The cavity diameter increased from 11 mm to 31 mm over 10 years,and a ground glass opacity lesion appeared around the bulla on computed tomography.A second operation confirmed that the lesion was lepidic predominant adenocarcinoma.Here we report a rare case of lung cancer developing from a focal bulla to a thinwalled adenocarcinoma for more than 10 years and confirm that the check-valve mechanism explains the pathogenesis.CONCLUSION Solitary thin-walled lung adenocarcinoma is a rare tumor in terms of its clinical manifestations,pathogenesis,and disease progression.The check-valve mechanism can explain the cause of thin-walled lung cancer.Close follow-up and accurate imaging are necessary.展开更多
There are lots of researches on fixture layout optimization for large thin-walled parts.Current researches focus on the positioning problem,i.e.,optimizing the positions of a constant number of fixtures.However,how to...There are lots of researches on fixture layout optimization for large thin-walled parts.Current researches focus on the positioning problem,i.e.,optimizing the positions of a constant number of fixtures.However,how to determine the number of fixtures is ignored.In most cases,the number of fixtures located on large thin-walled parts is determined based on engineering experience,which leads to huge fixture number and extra waste.Therefore,this paper constructs an optimization model to minimize the number of fixtures.The constraints are set in the optimization model to ensure that the part deformation is within the surface profile tolerance.In addition,the assembly gap between two parts is also controlled.To conduct the optimization,this paper develops an improved particle swarm optimization(IPSO)algorithm by integrating the shrinkage factor and adaptive inertia weight.In the algorithm,particles are encoded according to the fixture position.Each dimension of the particle is assigned to a sub-region by constraining the optional position range of each fixture to improve the optimization efficiency.Finally,a case study on ship curved panel assembly is provided to prove that our method can optimize the number of fixtures while meeting the assembly quality requirements.This research proposes a method to optimize the number of fixtures,which can reduce the number of fixtures and achieve deformation control at the same time.展开更多
Recently, demand for the lightweight alloy in electric/electronic housings has been greatly increased. However, among the lightweight alloys, aluminum alloy thin-walled die casting is problematic because it is quite d...Recently, demand for the lightweight alloy in electric/electronic housings has been greatly increased. However, among the lightweight alloys, aluminum alloy thin-walled die casting is problematic because it is quite difficult to achieve sufficient fluidity and feedability to fill the thin cavity as the wall thickness becomes less than 1mm. Therefore, in this study, thin-walled die casting of aluminum (Al-Si-Cu alloy: ALDC 12) in size of notebook computer housing and thickness of 0.8 mm was investigated by solidification simulation (MAGMA soft) and actual casting experiment (Buhler Evolution B 53D). Three different types of gating design, finger, tangential and split type with 6 vertical runners, were simulated and the results showed that sound thin-walled die casting was possible with tangential and split type gating design because those gates allowed aluminum melt to flow into the thin cavity uniformly and split type gating system was preferable gating design comparing to tangential type gating system at the point of view of soundness of casting and distortion generated after solidification. Also, the solidification simulation agreed well with the actual die-casting and the casting showed no casting defects and distortion.展开更多
An increasing number of researchers have researched fixture layout optimization for thin-walled part assembly during the past decades.However,few papers systematically review these researches.By analyzing existing lit...An increasing number of researchers have researched fixture layout optimization for thin-walled part assembly during the past decades.However,few papers systematically review these researches.By analyzing existing literature,this paper summarizes the process of fixture layout optimization and the methods applied.The process of optimization is made up of optimization objective setting,assembly variation/deformation modeling,and fixture layout optimization.This paper makes a review of the fixture layout for thin-walled parts according to these three steps.First,two different kinds of optimization objectives are introduced.Researchers usually consider in-plane variations or out-of-plane deformations when designing objectives.Then,modeling methods for assembly variation and deformation are divided into two categories:Mechanism-based and data-based methods.Several common methods are discussed respectively.After that,optimization algorithms are reviewed systematically.There are two kinds of optimization algorithms:Traditional nonlinear programming and heuristic algorithms.Finally,discussions on the current situation are provided.The research direction of fixture layout optimization in the future is discussed from three aspects:Objective setting,improving modeling accuracy and optimization algorithms.Also,a new research point for fixture layout optimization is discussed.This paper systematically reviews the research on fixture layout optimization for thin-walled parts,and provides a reference for future research in this field.展开更多
Lightweight thin-walled structures with lattice infill are widely desired in satellite for their high stiffness-to-weight ratio and superior buckling strength resulting fromthe sandwich effect.Such structures can be f...Lightweight thin-walled structures with lattice infill are widely desired in satellite for their high stiffness-to-weight ratio and superior buckling strength resulting fromthe sandwich effect.Such structures can be fabricated bymetallic additive manufacturing technique,such as selective laser melting(SLM).However,the maximum dimensions of actual structures are usually in a sub-meter scale,which results in restrictions on their appliance in aerospace and other fields.In this work,a meter-scale thin-walled structure with lattice infill is designed for the fuel tank supporting component of the satellite by integrating a self-supporting lattice into the thickness optimization of the thin-wall.The designed structure is fabricated by SLM of AlSi10Mg and cold metal transfer welding technique.Quasi-static mechanical tests and vibration tests are both conducted to verify the mechanical strength of the designed large-scale lattice thin-walled structure.The experimental results indicate that themeter-scale thin-walled structure with lattice infill could meet the dimension and lightweight requirements of most spacecrafts.展开更多
As critical components of aircraft skins and rocket fuel storage tank shells,large thin-walled workpieces are susceptible to vibration and deformation during machining due to their weak local stiffness.To address thes...As critical components of aircraft skins and rocket fuel storage tank shells,large thin-walled workpieces are susceptible to vibration and deformation during machining due to their weak local stiffness.To address these challenges,we propose a novel tunable electromagnetic semi-active dynamic vibration absorber(ESADVA),which integrates with a magnetic suction follower to form a followed ESADVA(follow-ESADVA)for mirror milling.This system combines a tunable magnet oscillator with a follower,enabling real-time vibration absorption and condition feedback throughout the milling process.Additionally,the device supports self-sensing and frequency adjustment by providing feedback to a linear actuator,which alters the distance between magnets.This resolves the traditional issue of being unable to directly monitor vibration at the machining point due to space constraints and tool interference.The frequency shift characteristics and vibration absorption performance are comprehensively investigated.Theoretical and experimental results demonstrate that the prototyped follow-ESADVA achieves frequency synchronization with the milling tool,resulting in a vibration suppression rate of approximately 47.57%.Moreover,the roughness of the machined surface decreases by18.95%,significantly enhancing the surface quality.The results of this work pave the way for higher-quality machined surfaces and a more stable mirror milling process.展开更多
Pitting corrosion is harmful during bridge construction,which will lead to uneven roughness of steel surfaces and reduce the thickness of steel.Hence,the effect of pitting corrosion on the mechanical properties of col...Pitting corrosion is harmful during bridge construction,which will lead to uneven roughness of steel surfaces and reduce the thickness of steel.Hence,the effect of pitting corrosion on the mechanical properties of cold-formed thin-walled steel stub columns is studied,and the empirical formulas are established through regression fitting to predict the ultimate load of web and flange under pitting corrosion.In detail,the failure modes and load-displacement curves of specimens with different locations,area ratios,and depths are obtained through a large number of non-linear finite element analysis.As for the specimens with pitting corrosion on the web,all the specimens are subject to local buckling failure,and the failure mode will not change with pitting corrosion,but the failure location will change with pitting corrosion location;the size,location,and area ratio of pitting corrosion have little influence on the ultimate load of cold-formed thin-walled steel short columns,but the loss rate of pitting corrosion section area has a greater impact on the ultimate bearing capacity.As for the specimen with flange pitting corrosion,the location and area ratio of pitting corrosion have less influence on the ultimate load of cold-formed thin-walled steel short columns,and the section area loss rate has greater influence on the ultimate bearing capacity;the impact of web pitting corrosion on the ultimate load is greater than that of flange pitting corrosion under the same condition of pitting corrosion section area.The prediction formulas of limit load which are suitable for pitting corrosion of web and flange are established,which can provide a reference for performance evaluation of corroded cold-formed thin-walled steel.展开更多
BACKGROUND Gallbladder neuroendocrine carcinoma(NEC)represents a subtype of gallbladder malignancies characterized by a low incidence,aggressive nature,and poor prognosis.Despite its clinical severity,the genetic alte...BACKGROUND Gallbladder neuroendocrine carcinoma(NEC)represents a subtype of gallbladder malignancies characterized by a low incidence,aggressive nature,and poor prognosis.Despite its clinical severity,the genetic alterations,mechanisms,and signaling pathways underlying gallbladder NEC remain unclear.CASE SUMMARY This case study presents a rare instance of primary gallbladder NEC in a 73-year-old female patient,who underwent a radical cholecystectomy with hepatic hilar lymphadenectomy and resection of liver segments IV-B and V.Targeted gene sequencing and bioinformatics analysis tools,including STRING,GeneMANIA,Metascape,TRRUST,Sangerbox,cBioPortal and GSCA,were used to analyze the biological functions and features of mutated genes in gallbladder NEC.Twelve mutations(APC,ARID2,IFNA6,KEAP1,RB1,SMAD4,TP53,BTK,GATA1,GNAS,and PRDM3)were identified,and the tumor mutation burden was determined to be 9.52 muts/Mb via targeted gene sequencing.A protein-protein interaction network showed significant interactions among the twelve mutated genes.Gene Ontology and Kyoto Encyclopedia of Genes and Genomes analyses were used to assess mutation functions and pathways.The results revealed 40 tumor-related pathways.A key regulatory factor for gallbladder NEC-related genes was identified,and its biological functions and features were compared with those of gallbladder carcinoma.CONCLUSION Gallbladder NEC requires standardized treatment.Comparisons with other gallbladder carcinomas revealed clinical phenotypes,molecular alterations,functional characteristics,and enriched pathways.展开更多
BACKGROUND Hydroxyurea,an antimetabolite,is frequently prescribed for various hemato-logical disorders,and its common side effects include gastrointestinal problems,cutaneous or mucosal lesions and pyrexia/fever.CASE ...BACKGROUND Hydroxyurea,an antimetabolite,is frequently prescribed for various hemato-logical disorders,and its common side effects include gastrointestinal problems,cutaneous or mucosal lesions and pyrexia/fever.CASE SUMMARY This study reports the case of a 67-year-old woman who developed recurrent abdominal pain after 10 years of continuous hydroxyurea therapy for primary thrombocythemia.Colonoscopy revealed an ileocecal ulcer.After discontinuing hydroxyurea therapy for 6 months,follow-up colonoscopy showed a significant reduction in the ulceration.CONCLUSION We consider cecal ulcers as a rare complication of hydroxyurea therapy which typically resolves upon stopping the drug.展开更多
BACKGROUND Gallbladder cancer(GBC)is the most common and aggressive subtype of biliary tract cancer(BTC)and has a poor prognosis.A newly developed regimen of gemcitabine,cisplatin,and durvalumab shows promise for the ...BACKGROUND Gallbladder cancer(GBC)is the most common and aggressive subtype of biliary tract cancer(BTC)and has a poor prognosis.A newly developed regimen of gemcitabine,cisplatin,and durvalumab shows promise for the treatment of advanced BTC.However,the efficacy of this treatment for GBC remains unclear.CASE SUMMARY In this report,we present a case in which the triple-drug regimen exhibited marked effectiveness in treating locally advanced GBC,thus leading to a long-term survival benefit.A 68-year-old man was diagnosed with locally advanced GBC,which rendered him ineligible for curative surgery.Following three cycles of therapy,a partial response was observed.After one year of combined therapy,a clinical complete response was successfully achieved.Subsequent maintenance therapy with durvalumab monotherapy resulted in a disease-free survival of 9 months for the patient.The patient experienced tolerable toxicities of reversible grade 2 nausea and fatigue.Tolerable adverse events were observed in the patient throughout the entirety of the treatment.CONCLUSION The combination of gemcitabine and cisplatin chemotherapy with durvalumab was proven to be an effective treatment approach for advanced GBC,with manageable adverse events.Further research is warranted to substantiate the effectiveness of the combined regimen in the context of GBC.展开更多
BACKGROUND Acute hyperglycemia due to insulin resistance is common in critically ill patients,typically managed with insulin infusion.However,the occurrence of transient extreme insulin resistance(EIR)requiring except...BACKGROUND Acute hyperglycemia due to insulin resistance is common in critically ill patients,typically managed with insulin infusion.However,the occurrence of transient extreme insulin resistance(EIR)requiring exceptional high-dose insulin is rare.CASE SUMMARY We present the case of a 68-year-old woman with pneumonia who suffered an out-of-hospital cardiac arrest,subsequently developing transient EIR following a new episode of sepsis.Remarkably,insulin resistance rapidly reversed when the insulin infusion rate peaked at 960 units/hour(a total of 18224 units on that day),and it was promptly titrated down to zero upon achieving the target glucose level.CONCLUSION Exceptional high-dose insulin infusion may be required in critically ill patients with stress-related EIR,which is typically transient.Clinicians should be aware of the phenomenon and cautious to avoid hypoglycemia and fluid overload during the steep titration of high-dose insulin infusion.展开更多
BACKGROUND Thrombophilia contributes to a significant increased risk of venous thromboembolism and can be either inherited or acquired.Hereditary thrombophilia may arise from various gene mutations,some of which have ...BACKGROUND Thrombophilia contributes to a significant increased risk of venous thromboembolism and can be either inherited or acquired.Hereditary thrombophilia may arise from various gene mutations,some of which have not even been adequately reported or poorly understood.Previous studies reported a rare and novel missense mutation in the prothrombin gene(p.Arg596Gln),known as prothrombin Belgrade.The mechanisms and therapeutic strategies associated with prothrombin Belgrade mutation have not been fully elucidated.CASE SUMMARY We present the case of a 26-year-old woman with recurrent systemic thrombosis induced by prothrombin Belgrade mutation.The patient suffered from cerebral venous sinus thrombosis that rapidly progressed to systemic thrombosis,alongside a family history of cerebral thrombosis,and no traditional risk factors or abnormal coagulation function.Whole-genome sequencing detected a novel and rare heterozygous prothrombin missense mutation,c.1787G>T(p.Arg596Gln),which was responsible for the major etiology of the systemic thrombosis.CONCLUSION This case strengthens our understanding about hereditary basis of thrombophilia and provokes considerations for therapeutic options on prothrombin Belgrade mutation.展开更多
BACKGROUND Acinic cell carcinoma(ACC)is a malignant epithelial neoplasm that commonly occurs in the parotid gland.It is known to have a high recurrence rate and the potential to metastasize to the lung or cervical lym...BACKGROUND Acinic cell carcinoma(ACC)is a malignant epithelial neoplasm that commonly occurs in the parotid gland.It is known to have a high recurrence rate and the potential to metastasize to the lung or cervical lymph nodes.However,few cases of ACC with bone metastasis have been reported in the medical literature.CASE SUMMARY The clinical significance of this case report lies in the unique site of occurrence of the metastasis:To the best of our knowledge,this report is the only literature documenting ACC arising in a shoulder mass.CONCLUSION Unusual presentations of uncommon malignancies can present diagnostic challenges for both surgeons and histopathologists.It is important to be aware of these rare occurrences in order to provide the best possible treatment for patients.展开更多
BACKGROUND The liver is the most common site of digestive system tumor metastasis,but not all liver metastases can be traced back to the primary lesions.Although it is unusual,syphilis can impact the liver,manifesting...BACKGROUND The liver is the most common site of digestive system tumor metastasis,but not all liver metastases can be traced back to the primary lesions.Although it is unusual,syphilis can impact the liver,manifesting as syphilitic hepatitis with inflammatory nodules,which might be misdiagnosed as metastasis.CASE SUMMARY This case report involves a 46-year-old female who developed right upper abdominal pain and intermittent low fever that persisted for more than three months.No definitive diagnosis of a tumor had been made in the past decades,but signs of multiple liver metastases were recognized after a computed tomo-graphy scan without evidence of primary lesions.With positive serological tests for syphilis and a biopsy of the liver nodules,a diagnosis of hepatic syphilis was made and confirmed with follow-up nodule reduction after anti-syphilis therapy.CONCLUSION Clinicians must be aware of the possibility that syphilis can cause hepatic inflam-matory masses,especially when liver metastasis is suspected without evidence of primary lesions.A definitive diagnosis should be established in conjunction with a review of the patient’s medical history for accurate therapeutic intervention.展开更多
BACKGROUND Congenital cytomegalovirus(CMV)infection represents a significant public health concern as the most prevalent viral infection in newborns,potentially leading to severe neurological and developmental complic...BACKGROUND Congenital cytomegalovirus(CMV)infection represents a significant public health concern as the most prevalent viral infection in newborns,potentially leading to severe neurological and developmental complications.The majority of cases are asymptomatic and remain undetected during pregnancy due to the absence of effective screening methods.CASE SUMMARY A 27-year-old primigravida presented for early pregnancy ultrasound,which revealed an atypical finding:A normal anechoic thalamus appearing hyperechoic on the mid-sagittal view of the fetal head.Subsequent ultrasound examinations during mid and late gestation demonstrated classic intracranial features sug-gestive of congenital CMV infection.Chromosomal karyotyping and microarray analysis of the fetus yielded no significant abnormalities.Following compre-hensive prenatal counseling regarding potential adverse fetal outcomes,the patient elected to continue her pregnancy.She ultimately underwent cesarean delivery at 42 weeks gestation at another facility,resulting in the birth of a female neonate.At five months of age,the infant presented with epilepsy and significant growth and developmental delays.CONCLUSION Congenital CMV infection occurs during the first trimester may manifest as hyperechoic thalamus which can be revealed by ultrasound in the mid-saggital view of the fetal head.Future research should investigate the correlation between echogenic thalamus and developmental outcomes,as well as explore early sc-reening techniques for suspected congenital CMV infection cases.展开更多
BACKGROUND Cleidocranial dysplasia(CCD)is an infrequent clinical condition with an autosomal dominant inheritance pattern.It is characterized by abnormal clavicles,patent sutures and fontanelles,supernumerary teeth,an...BACKGROUND Cleidocranial dysplasia(CCD)is an infrequent clinical condition with an autosomal dominant inheritance pattern.It is characterized by abnormal clavicles,patent sutures and fontanelles,supernumerary teeth,and short stature.Approximately 60%-70%of patients with CCD have mutations in the RUNX family transcription factor 2 gene.However,prenatal diagnosis of CCD is difficult when the family history is unknown.CASE SUMMARY We report a rare case of fetal CCD with an unknown family history,confirmed by prenatal ultrasonography and genetic testing at a gestational age of 16 weeks.The genetic reports indicated that the fetus carried pathogenic mutations in the RUNX family transcription factor 2 gene(c.674G>A).After careful consideration,the pregnant woman and her family decided to continue the pregnancy.CONCLUSION Definitive prenatal diagnosis of CCD should include family history,ultrasound diagnosis,and genetic analysis,especially if family history is unknown.展开更多
BACKGROUND Acute pancreatitis in pregnancy is a rare but serious condition that can lead to high maternal mortality and fetal loss.Instances of pregnancy complicated by severe acute pancreatitis,particularly with subs...BACKGROUND Acute pancreatitis in pregnancy is a rare but serious condition that can lead to high maternal mortality and fetal loss.Instances of pregnancy complicated by severe acute pancreatitis,particularly with subsequent respiratory and cardiac arrest,are rarely reported.CASE SUMMARY We present the case of a 35-year-old woman,at 36+5 weeks of gestation,who presented with paroxysmal epigastric pain accompanied by low back pain,nausea,and vomiting.According to the clinical symptoms,B-ultrasound imaging and biochemical indicators,the patient was diagnosed with acute pancreatitis and initially managed conservatively.However,3 hours after admission,the patient experienced respiratory and cardiac arrest,and the fetus died.In this case,the adverse outcomes occurred due to the lack of aggressive fluid resuscitation and an active surgical intervention.CONCLUSION Implementing aggressive fluid resuscitation to sustain tissue perfusion,alongside the proactive evaluation of pharmacological agents that suppress gastric acid secretion and inhibit pancreatic enzyme activity,may be beneficial in mitigating the risk of a severely adverse prognosis.Effective management of acute pancreatitis during pregnancy requires careful timing of surgical intervention,a thorough evaluation of the risks and benefits regarding the continuation or termination of pregnancy,and a focus on safeguarding both maternal and fetal health.展开更多
BACKGROUND Ependymoma with lipomatous differentiation is a rare type of ependymoma.The ZFTA fusion-positive supratentorial ependymoma is a novel tumor type in the 2021 World Health Organization classification of centr...BACKGROUND Ependymoma with lipomatous differentiation is a rare type of ependymoma.The ZFTA fusion-positive supratentorial ependymoma is a novel tumor type in the 2021 World Health Organization classification of central nervous system tumors.ZFTA fusion-positive lipomatous ependymoma has not been reported to date.CASE SUMMARY We reported a case of a 15-year-old Chinese male who had a sudden convulsion lasting approximately six minutes.Magnetic resonance imaging showed a round cystic shadow of approximately 1.9 cm×1.5 cm×1.9 cm under the right parieto-occipital cortex.Microscopic examination showed characteristic perivascular pseudorosettes and adipose differentiation in the cytoplasm.Immunohisto-chemical staining showed that the tumor cells were negative for cytokeratin,NeuN,Syn and p53,but positive for GFAP,vimentin and S-100 protein.Signi-ficant punctate intracytoplasmic EMA immunoreactivity was observed.The level of Ki-67 was about 5%.Genetic analysis revealed ZFTA:RELA fusion.A cranio-tomy with total excision of the tumor was performed.The follow-up time was 36 months,no evidence of disease recurrence was found in magnetic resonance imaging.CONCLUSION Based on these findings,the patient was diagnosed as a ependymoma with ZFTA fusion and lipomatous differentiation.This case report provides information on the microscopic morphological features of ependymoma with ZFTA fusion and lipomatous differentiation,which can help pathologists to make a definitive diagnosis of this tumor.展开更多
BACKGROUND Cervical cancer is a formidable global health issue,particularly affecting women in lower-middle-income countries with little or no access to preventative vaccines,screening programs,and treatment modalitie...BACKGROUND Cervical cancer is a formidable global health issue,particularly affecting women in lower-middle-income countries with little or no access to preventative vaccines,screening programs,and treatment modalities.The case report presents a unique case of a large cervical cancer achieving complete response(CR)with concurrent chemoradiotherapy(CCRT),highlighting the effectiveness of this treatment approach even in advanced stages and underscoring the importance of adaptive radiotherapy(RT)in optimizing patient outcomes.CASE SUMMARY We present the case of a 53-year-old woman who presented with four years of abnormal vaginal bleeding and was found to have p16-positive,moderately differentiated cervical squamous cell carcinoma.The tumor measured 14 cm×12 cm×8 cm,the largest size reported in the literature to achieve CR with CCRT.Despite this monumental feat,the patient remained disease-free and is currently on follow-up for 2 years;however,she continued to suffer from substantial morbidity caused by a vesicovaginal fistula and hydronephrosis,underscoring the continuing impact of cervical cancer on quality of life.CONCLUSION In this case report,we highlight the effectiveness of CCRT in achieving CR,even in cases of bulky cervical cancer,with adaptive RT offering a customized strategy to improve patient outcomes.We also emphasize the necessity for multidisciplinary team discussions and highlight the need for strategies to mitigate treatment-related toxicities and long-term complications.展开更多
BACKGROUND Wilson's disease(WD)is a rare metabolic disorder of copper accumulation in organs such as liver,brain,and cornea.Diagnoses and treatments are challenging in settings,where advanced diagnostic tests are ...BACKGROUND Wilson's disease(WD)is a rare metabolic disorder of copper accumulation in organs such as liver,brain,and cornea.Diagnoses and treatments are challenging in settings,where advanced diagnostic tests are unavailable,copper chelating agents are frequently scarce,healthcare professionals lack disease awareness,and medical follow-ups are limited.Prompt diagnoses and treatments help prevent complications,improve patients’quality of life,and ensure a normal life expectancy.The clinical presentations and outcomes of WD can vary within a single family.CASE SUMMARY We present the cases of two siblings(19 and 27 years)from a consanguineous family in rural Ecuador,diagnosed as having WD during a family screening.The male patient,diagnosed at age 19 after his brother’s death from acute liver failure,presented with compensated cirrhosis,neurological symptoms,and bilateral Kayser-Fleischer rings.He developed progressive neurological deterioration during an irregular treatment with D-penicillamine due to medication shortages.His condition improved upon switching to trientine tetrahydrochloride,and his neurological symptoms improved over an 8-year period of follow-ups.The female patient,diagnosed at age 10,exhibited only biochemical alterations.Her treatment history was similar;however,she remained asymptomatic without disease progression over the same follow-up period.We discuss the potential influence of epigenetic mechanisms and modifier genes on the various phenotypes,emphasizing the need for research in these areas to optimize therapeutic strategies.CONCLUSION Our patients’medical histories show how early diagnosis and treatment can prevent disease progression;and,how suboptimal treatments impact disease outcomes.展开更多
文摘BACKGROUND Thin-walled lung cancer manifests as a cystic lesion,mostly adenocarcinoma.It is often misdiagnosed as a benign lesion in clinical practice,thus delaying the diagnosis and surgical treatment.Its natural course is rarely recorded and observed;thus,the pathogenesis and diagnosis need to be clarified and improved.CASE SUMMARY A 66-year-old man developed a mass in the upper lobe of the right lung and a small,thin-walled cavity in the lower lobe of the right lung in 2007.The right upper lobe mass was confirmed to be adenocarcinoma after surgery.The cavity diameter increased from 11 mm to 31 mm over 10 years,and a ground glass opacity lesion appeared around the bulla on computed tomography.A second operation confirmed that the lesion was lepidic predominant adenocarcinoma.Here we report a rare case of lung cancer developing from a focal bulla to a thinwalled adenocarcinoma for more than 10 years and confirm that the check-valve mechanism explains the pathogenesis.CONCLUSION Solitary thin-walled lung adenocarcinoma is a rare tumor in terms of its clinical manifestations,pathogenesis,and disease progression.The check-valve mechanism can explain the cause of thin-walled lung cancer.Close follow-up and accurate imaging are necessary.
基金Supported by National Natural Science Foundation of China(Grant No.52005371)Shanghai Pujiang Program of China(Grant No.2020PJD071)+1 种基金Shanghai Municipal Natural Science Foundation of China(Grant No.22ZR1463900)Fundamental Research Funds for the Central Universities of China.
文摘There are lots of researches on fixture layout optimization for large thin-walled parts.Current researches focus on the positioning problem,i.e.,optimizing the positions of a constant number of fixtures.However,how to determine the number of fixtures is ignored.In most cases,the number of fixtures located on large thin-walled parts is determined based on engineering experience,which leads to huge fixture number and extra waste.Therefore,this paper constructs an optimization model to minimize the number of fixtures.The constraints are set in the optimization model to ensure that the part deformation is within the surface profile tolerance.In addition,the assembly gap between two parts is also controlled.To conduct the optimization,this paper develops an improved particle swarm optimization(IPSO)algorithm by integrating the shrinkage factor and adaptive inertia weight.In the algorithm,particles are encoded according to the fixture position.Each dimension of the particle is assigned to a sub-region by constraining the optional position range of each fixture to improve the optimization efficiency.Finally,a case study on ship curved panel assembly is provided to prove that our method can optimize the number of fixtures while meeting the assembly quality requirements.This research proposes a method to optimize the number of fixtures,which can reduce the number of fixtures and achieve deformation control at the same time.
文摘Recently, demand for the lightweight alloy in electric/electronic housings has been greatly increased. However, among the lightweight alloys, aluminum alloy thin-walled die casting is problematic because it is quite difficult to achieve sufficient fluidity and feedability to fill the thin cavity as the wall thickness becomes less than 1mm. Therefore, in this study, thin-walled die casting of aluminum (Al-Si-Cu alloy: ALDC 12) in size of notebook computer housing and thickness of 0.8 mm was investigated by solidification simulation (MAGMA soft) and actual casting experiment (Buhler Evolution B 53D). Three different types of gating design, finger, tangential and split type with 6 vertical runners, were simulated and the results showed that sound thin-walled die casting was possible with tangential and split type gating design because those gates allowed aluminum melt to flow into the thin cavity uniformly and split type gating system was preferable gating design comparing to tangential type gating system at the point of view of soundness of casting and distortion generated after solidification. Also, the solidification simulation agreed well with the actual die-casting and the casting showed no casting defects and distortion.
基金Supported by National Natural Science Foundation of China(Grant No.52005371)Shanghai Municipal Natural Science Foundation of China(Grant No.22ZR1463900)+1 种基金Fundamental Research Funds for the Central Universities of China(Grant No.22120220649)State Key Laboratory of Mechanical System and Vibration of China(Grant No.MSV202318).
文摘An increasing number of researchers have researched fixture layout optimization for thin-walled part assembly during the past decades.However,few papers systematically review these researches.By analyzing existing literature,this paper summarizes the process of fixture layout optimization and the methods applied.The process of optimization is made up of optimization objective setting,assembly variation/deformation modeling,and fixture layout optimization.This paper makes a review of the fixture layout for thin-walled parts according to these three steps.First,two different kinds of optimization objectives are introduced.Researchers usually consider in-plane variations or out-of-plane deformations when designing objectives.Then,modeling methods for assembly variation and deformation are divided into two categories:Mechanism-based and data-based methods.Several common methods are discussed respectively.After that,optimization algorithms are reviewed systematically.There are two kinds of optimization algorithms:Traditional nonlinear programming and heuristic algorithms.Finally,discussions on the current situation are provided.The research direction of fixture layout optimization in the future is discussed from three aspects:Objective setting,improving modeling accuracy and optimization algorithms.Also,a new research point for fixture layout optimization is discussed.This paper systematically reviews the research on fixture layout optimization for thin-walled parts,and provides a reference for future research in this field.
基金The authors are grateful for the support by National Key Research and Development Program of China(2021YFF0500300,2020YFB1708300)the National Natural Science Foundation of China(52205280,12172041).
文摘Lightweight thin-walled structures with lattice infill are widely desired in satellite for their high stiffness-to-weight ratio and superior buckling strength resulting fromthe sandwich effect.Such structures can be fabricated bymetallic additive manufacturing technique,such as selective laser melting(SLM).However,the maximum dimensions of actual structures are usually in a sub-meter scale,which results in restrictions on their appliance in aerospace and other fields.In this work,a meter-scale thin-walled structure with lattice infill is designed for the fuel tank supporting component of the satellite by integrating a self-supporting lattice into the thickness optimization of the thin-wall.The designed structure is fabricated by SLM of AlSi10Mg and cold metal transfer welding technique.Quasi-static mechanical tests and vibration tests are both conducted to verify the mechanical strength of the designed large-scale lattice thin-walled structure.The experimental results indicate that themeter-scale thin-walled structure with lattice infill could meet the dimension and lightweight requirements of most spacecrafts.
基金Project supported by the National Natural Science Foundation of China(Nos.12172248,12021002,12302022,and 12132010)the Tianjin Research Program of Application Foundation and Advanced Technology of China(No.22JCQNJC00780)IoT Standards and Application Key Laboratory of the Ministry of Industry and Information Technology of China(No.202306)。
文摘As critical components of aircraft skins and rocket fuel storage tank shells,large thin-walled workpieces are susceptible to vibration and deformation during machining due to their weak local stiffness.To address these challenges,we propose a novel tunable electromagnetic semi-active dynamic vibration absorber(ESADVA),which integrates with a magnetic suction follower to form a followed ESADVA(follow-ESADVA)for mirror milling.This system combines a tunable magnet oscillator with a follower,enabling real-time vibration absorption and condition feedback throughout the milling process.Additionally,the device supports self-sensing and frequency adjustment by providing feedback to a linear actuator,which alters the distance between magnets.This resolves the traditional issue of being unable to directly monitor vibration at the machining point due to space constraints and tool interference.The frequency shift characteristics and vibration absorption performance are comprehensively investigated.Theoretical and experimental results demonstrate that the prototyped follow-ESADVA achieves frequency synchronization with the milling tool,resulting in a vibration suppression rate of approximately 47.57%.Moreover,the roughness of the machined surface decreases by18.95%,significantly enhancing the surface quality.The results of this work pave the way for higher-quality machined surfaces and a more stable mirror milling process.
基金funded by the‘Research Project of the Sucheng to Sihong Section of the Yanluo Expressway-Measurement Technology and Application of Bridge Quality Project Based on UAV Binocular Imaging(No.00-00-JSFW-20230203-029)’,received by H.Z.Wang.
文摘Pitting corrosion is harmful during bridge construction,which will lead to uneven roughness of steel surfaces and reduce the thickness of steel.Hence,the effect of pitting corrosion on the mechanical properties of cold-formed thin-walled steel stub columns is studied,and the empirical formulas are established through regression fitting to predict the ultimate load of web and flange under pitting corrosion.In detail,the failure modes and load-displacement curves of specimens with different locations,area ratios,and depths are obtained through a large number of non-linear finite element analysis.As for the specimens with pitting corrosion on the web,all the specimens are subject to local buckling failure,and the failure mode will not change with pitting corrosion,but the failure location will change with pitting corrosion location;the size,location,and area ratio of pitting corrosion have little influence on the ultimate load of cold-formed thin-walled steel short columns,but the loss rate of pitting corrosion section area has a greater impact on the ultimate bearing capacity.As for the specimen with flange pitting corrosion,the location and area ratio of pitting corrosion have less influence on the ultimate load of cold-formed thin-walled steel short columns,and the section area loss rate has greater influence on the ultimate bearing capacity;the impact of web pitting corrosion on the ultimate load is greater than that of flange pitting corrosion under the same condition of pitting corrosion section area.The prediction formulas of limit load which are suitable for pitting corrosion of web and flange are established,which can provide a reference for performance evaluation of corroded cold-formed thin-walled steel.
基金Supported by School-Level Key Projects at Bengbu Medical College,No.2021byzd109.
文摘BACKGROUND Gallbladder neuroendocrine carcinoma(NEC)represents a subtype of gallbladder malignancies characterized by a low incidence,aggressive nature,and poor prognosis.Despite its clinical severity,the genetic alterations,mechanisms,and signaling pathways underlying gallbladder NEC remain unclear.CASE SUMMARY This case study presents a rare instance of primary gallbladder NEC in a 73-year-old female patient,who underwent a radical cholecystectomy with hepatic hilar lymphadenectomy and resection of liver segments IV-B and V.Targeted gene sequencing and bioinformatics analysis tools,including STRING,GeneMANIA,Metascape,TRRUST,Sangerbox,cBioPortal and GSCA,were used to analyze the biological functions and features of mutated genes in gallbladder NEC.Twelve mutations(APC,ARID2,IFNA6,KEAP1,RB1,SMAD4,TP53,BTK,GATA1,GNAS,and PRDM3)were identified,and the tumor mutation burden was determined to be 9.52 muts/Mb via targeted gene sequencing.A protein-protein interaction network showed significant interactions among the twelve mutated genes.Gene Ontology and Kyoto Encyclopedia of Genes and Genomes analyses were used to assess mutation functions and pathways.The results revealed 40 tumor-related pathways.A key regulatory factor for gallbladder NEC-related genes was identified,and its biological functions and features were compared with those of gallbladder carcinoma.CONCLUSION Gallbladder NEC requires standardized treatment.Comparisons with other gallbladder carcinomas revealed clinical phenotypes,molecular alterations,functional characteristics,and enriched pathways.
基金Supported by National Natural Science Foundation of China,No.82370604Major Science and Technology innovation Project of Fujian Province,No.2023Y9269Natural Science Foundation of Fujian Province,China,No.2023J01239.
文摘BACKGROUND Hydroxyurea,an antimetabolite,is frequently prescribed for various hemato-logical disorders,and its common side effects include gastrointestinal problems,cutaneous or mucosal lesions and pyrexia/fever.CASE SUMMARY This study reports the case of a 67-year-old woman who developed recurrent abdominal pain after 10 years of continuous hydroxyurea therapy for primary thrombocythemia.Colonoscopy revealed an ileocecal ulcer.After discontinuing hydroxyurea therapy for 6 months,follow-up colonoscopy showed a significant reduction in the ulceration.CONCLUSION We consider cecal ulcers as a rare complication of hydroxyurea therapy which typically resolves upon stopping the drug.
基金Supported by General Project of Natural Science Foundation of Chongqing,China,No.cstc2021jcyj-msxmX0604Chongqing Doctoral"Through Train"Research Program,China,No.CSTB2022BSXM-JCX0045.
文摘BACKGROUND Gallbladder cancer(GBC)is the most common and aggressive subtype of biliary tract cancer(BTC)and has a poor prognosis.A newly developed regimen of gemcitabine,cisplatin,and durvalumab shows promise for the treatment of advanced BTC.However,the efficacy of this treatment for GBC remains unclear.CASE SUMMARY In this report,we present a case in which the triple-drug regimen exhibited marked effectiveness in treating locally advanced GBC,thus leading to a long-term survival benefit.A 68-year-old man was diagnosed with locally advanced GBC,which rendered him ineligible for curative surgery.Following three cycles of therapy,a partial response was observed.After one year of combined therapy,a clinical complete response was successfully achieved.Subsequent maintenance therapy with durvalumab monotherapy resulted in a disease-free survival of 9 months for the patient.The patient experienced tolerable toxicities of reversible grade 2 nausea and fatigue.Tolerable adverse events were observed in the patient throughout the entirety of the treatment.CONCLUSION The combination of gemcitabine and cisplatin chemotherapy with durvalumab was proven to be an effective treatment approach for advanced GBC,with manageable adverse events.Further research is warranted to substantiate the effectiveness of the combined regimen in the context of GBC.
文摘BACKGROUND Acute hyperglycemia due to insulin resistance is common in critically ill patients,typically managed with insulin infusion.However,the occurrence of transient extreme insulin resistance(EIR)requiring exceptional high-dose insulin is rare.CASE SUMMARY We present the case of a 68-year-old woman with pneumonia who suffered an out-of-hospital cardiac arrest,subsequently developing transient EIR following a new episode of sepsis.Remarkably,insulin resistance rapidly reversed when the insulin infusion rate peaked at 960 units/hour(a total of 18224 units on that day),and it was promptly titrated down to zero upon achieving the target glucose level.CONCLUSION Exceptional high-dose insulin infusion may be required in critically ill patients with stress-related EIR,which is typically transient.Clinicians should be aware of the phenomenon and cautious to avoid hypoglycemia and fluid overload during the steep titration of high-dose insulin infusion.
文摘BACKGROUND Thrombophilia contributes to a significant increased risk of venous thromboembolism and can be either inherited or acquired.Hereditary thrombophilia may arise from various gene mutations,some of which have not even been adequately reported or poorly understood.Previous studies reported a rare and novel missense mutation in the prothrombin gene(p.Arg596Gln),known as prothrombin Belgrade.The mechanisms and therapeutic strategies associated with prothrombin Belgrade mutation have not been fully elucidated.CASE SUMMARY We present the case of a 26-year-old woman with recurrent systemic thrombosis induced by prothrombin Belgrade mutation.The patient suffered from cerebral venous sinus thrombosis that rapidly progressed to systemic thrombosis,alongside a family history of cerebral thrombosis,and no traditional risk factors or abnormal coagulation function.Whole-genome sequencing detected a novel and rare heterozygous prothrombin missense mutation,c.1787G>T(p.Arg596Gln),which was responsible for the major etiology of the systemic thrombosis.CONCLUSION This case strengthens our understanding about hereditary basis of thrombophilia and provokes considerations for therapeutic options on prothrombin Belgrade mutation.
文摘BACKGROUND Acinic cell carcinoma(ACC)is a malignant epithelial neoplasm that commonly occurs in the parotid gland.It is known to have a high recurrence rate and the potential to metastasize to the lung or cervical lymph nodes.However,few cases of ACC with bone metastasis have been reported in the medical literature.CASE SUMMARY The clinical significance of this case report lies in the unique site of occurrence of the metastasis:To the best of our knowledge,this report is the only literature documenting ACC arising in a shoulder mass.CONCLUSION Unusual presentations of uncommon malignancies can present diagnostic challenges for both surgeons and histopathologists.It is important to be aware of these rare occurrences in order to provide the best possible treatment for patients.
文摘BACKGROUND The liver is the most common site of digestive system tumor metastasis,but not all liver metastases can be traced back to the primary lesions.Although it is unusual,syphilis can impact the liver,manifesting as syphilitic hepatitis with inflammatory nodules,which might be misdiagnosed as metastasis.CASE SUMMARY This case report involves a 46-year-old female who developed right upper abdominal pain and intermittent low fever that persisted for more than three months.No definitive diagnosis of a tumor had been made in the past decades,but signs of multiple liver metastases were recognized after a computed tomo-graphy scan without evidence of primary lesions.With positive serological tests for syphilis and a biopsy of the liver nodules,a diagnosis of hepatic syphilis was made and confirmed with follow-up nodule reduction after anti-syphilis therapy.CONCLUSION Clinicians must be aware of the possibility that syphilis can cause hepatic inflam-matory masses,especially when liver metastasis is suspected without evidence of primary lesions.A definitive diagnosis should be established in conjunction with a review of the patient’s medical history for accurate therapeutic intervention.
文摘BACKGROUND Congenital cytomegalovirus(CMV)infection represents a significant public health concern as the most prevalent viral infection in newborns,potentially leading to severe neurological and developmental complications.The majority of cases are asymptomatic and remain undetected during pregnancy due to the absence of effective screening methods.CASE SUMMARY A 27-year-old primigravida presented for early pregnancy ultrasound,which revealed an atypical finding:A normal anechoic thalamus appearing hyperechoic on the mid-sagittal view of the fetal head.Subsequent ultrasound examinations during mid and late gestation demonstrated classic intracranial features sug-gestive of congenital CMV infection.Chromosomal karyotyping and microarray analysis of the fetus yielded no significant abnormalities.Following compre-hensive prenatal counseling regarding potential adverse fetal outcomes,the patient elected to continue her pregnancy.She ultimately underwent cesarean delivery at 42 weeks gestation at another facility,resulting in the birth of a female neonate.At five months of age,the infant presented with epilepsy and significant growth and developmental delays.CONCLUSION Congenital CMV infection occurs during the first trimester may manifest as hyperechoic thalamus which can be revealed by ultrasound in the mid-saggital view of the fetal head.Future research should investigate the correlation between echogenic thalamus and developmental outcomes,as well as explore early sc-reening techniques for suspected congenital CMV infection cases.
基金Supported by Science and Technology Development Plan Project of Weifang,No.2023YX005。
文摘BACKGROUND Cleidocranial dysplasia(CCD)is an infrequent clinical condition with an autosomal dominant inheritance pattern.It is characterized by abnormal clavicles,patent sutures and fontanelles,supernumerary teeth,and short stature.Approximately 60%-70%of patients with CCD have mutations in the RUNX family transcription factor 2 gene.However,prenatal diagnosis of CCD is difficult when the family history is unknown.CASE SUMMARY We report a rare case of fetal CCD with an unknown family history,confirmed by prenatal ultrasonography and genetic testing at a gestational age of 16 weeks.The genetic reports indicated that the fetus carried pathogenic mutations in the RUNX family transcription factor 2 gene(c.674G>A).After careful consideration,the pregnant woman and her family decided to continue the pregnancy.CONCLUSION Definitive prenatal diagnosis of CCD should include family history,ultrasound diagnosis,and genetic analysis,especially if family history is unknown.
文摘BACKGROUND Acute pancreatitis in pregnancy is a rare but serious condition that can lead to high maternal mortality and fetal loss.Instances of pregnancy complicated by severe acute pancreatitis,particularly with subsequent respiratory and cardiac arrest,are rarely reported.CASE SUMMARY We present the case of a 35-year-old woman,at 36+5 weeks of gestation,who presented with paroxysmal epigastric pain accompanied by low back pain,nausea,and vomiting.According to the clinical symptoms,B-ultrasound imaging and biochemical indicators,the patient was diagnosed with acute pancreatitis and initially managed conservatively.However,3 hours after admission,the patient experienced respiratory and cardiac arrest,and the fetus died.In this case,the adverse outcomes occurred due to the lack of aggressive fluid resuscitation and an active surgical intervention.CONCLUSION Implementing aggressive fluid resuscitation to sustain tissue perfusion,alongside the proactive evaluation of pharmacological agents that suppress gastric acid secretion and inhibit pancreatic enzyme activity,may be beneficial in mitigating the risk of a severely adverse prognosis.Effective management of acute pancreatitis during pregnancy requires careful timing of surgical intervention,a thorough evaluation of the risks and benefits regarding the continuation or termination of pregnancy,and a focus on safeguarding both maternal and fetal health.
文摘BACKGROUND Ependymoma with lipomatous differentiation is a rare type of ependymoma.The ZFTA fusion-positive supratentorial ependymoma is a novel tumor type in the 2021 World Health Organization classification of central nervous system tumors.ZFTA fusion-positive lipomatous ependymoma has not been reported to date.CASE SUMMARY We reported a case of a 15-year-old Chinese male who had a sudden convulsion lasting approximately six minutes.Magnetic resonance imaging showed a round cystic shadow of approximately 1.9 cm×1.5 cm×1.9 cm under the right parieto-occipital cortex.Microscopic examination showed characteristic perivascular pseudorosettes and adipose differentiation in the cytoplasm.Immunohisto-chemical staining showed that the tumor cells were negative for cytokeratin,NeuN,Syn and p53,but positive for GFAP,vimentin and S-100 protein.Signi-ficant punctate intracytoplasmic EMA immunoreactivity was observed.The level of Ki-67 was about 5%.Genetic analysis revealed ZFTA:RELA fusion.A cranio-tomy with total excision of the tumor was performed.The follow-up time was 36 months,no evidence of disease recurrence was found in magnetic resonance imaging.CONCLUSION Based on these findings,the patient was diagnosed as a ependymoma with ZFTA fusion and lipomatous differentiation.This case report provides information on the microscopic morphological features of ependymoma with ZFTA fusion and lipomatous differentiation,which can help pathologists to make a definitive diagnosis of this tumor.
文摘BACKGROUND Cervical cancer is a formidable global health issue,particularly affecting women in lower-middle-income countries with little or no access to preventative vaccines,screening programs,and treatment modalities.The case report presents a unique case of a large cervical cancer achieving complete response(CR)with concurrent chemoradiotherapy(CCRT),highlighting the effectiveness of this treatment approach even in advanced stages and underscoring the importance of adaptive radiotherapy(RT)in optimizing patient outcomes.CASE SUMMARY We present the case of a 53-year-old woman who presented with four years of abnormal vaginal bleeding and was found to have p16-positive,moderately differentiated cervical squamous cell carcinoma.The tumor measured 14 cm×12 cm×8 cm,the largest size reported in the literature to achieve CR with CCRT.Despite this monumental feat,the patient remained disease-free and is currently on follow-up for 2 years;however,she continued to suffer from substantial morbidity caused by a vesicovaginal fistula and hydronephrosis,underscoring the continuing impact of cervical cancer on quality of life.CONCLUSION In this case report,we highlight the effectiveness of CCRT in achieving CR,even in cases of bulky cervical cancer,with adaptive RT offering a customized strategy to improve patient outcomes.We also emphasize the necessity for multidisciplinary team discussions and highlight the need for strategies to mitigate treatment-related toxicities and long-term complications.
文摘BACKGROUND Wilson's disease(WD)is a rare metabolic disorder of copper accumulation in organs such as liver,brain,and cornea.Diagnoses and treatments are challenging in settings,where advanced diagnostic tests are unavailable,copper chelating agents are frequently scarce,healthcare professionals lack disease awareness,and medical follow-ups are limited.Prompt diagnoses and treatments help prevent complications,improve patients’quality of life,and ensure a normal life expectancy.The clinical presentations and outcomes of WD can vary within a single family.CASE SUMMARY We present the cases of two siblings(19 and 27 years)from a consanguineous family in rural Ecuador,diagnosed as having WD during a family screening.The male patient,diagnosed at age 19 after his brother’s death from acute liver failure,presented with compensated cirrhosis,neurological symptoms,and bilateral Kayser-Fleischer rings.He developed progressive neurological deterioration during an irregular treatment with D-penicillamine due to medication shortages.His condition improved upon switching to trientine tetrahydrochloride,and his neurological symptoms improved over an 8-year period of follow-ups.The female patient,diagnosed at age 10,exhibited only biochemical alterations.Her treatment history was similar;however,she remained asymptomatic without disease progression over the same follow-up period.We discuss the potential influence of epigenetic mechanisms and modifier genes on the various phenotypes,emphasizing the need for research in these areas to optimize therapeutic strategies.CONCLUSION Our patients’medical histories show how early diagnosis and treatment can prevent disease progression;and,how suboptimal treatments impact disease outcomes.