Search results for " Defects"

showing 10 items of 294 documents

Cardiac regenerative capacity is age- and disease-dependent in childhood heart disease

2018

Objective We sought to define the intrinsic stem cell capacity in pediatric heart lesions, and the effects of diagnosis and of age, in order to inform evidence-based use of potential autologous stem cell sources for regenerative medicine therapy. Methods Ventricular explants derived from patients with hypoplastic left heart syndrome (HLHS), tetralogy of Fallot (TF), dilated cardiomyopathy (DCM) and ventricular septal defect (VSD) were analyzed following standard in vitro culture conditions, which yielded cardiospheres (C-spheres), indicative of endogenous stem cell capacity. C-sphere counts generated per 5 mm3 tissue explant and the presence of cardiac progenitor cells were correlated to pa…

0301 basic medicineHeart Septal Defects VentricularAgingHeart diseaseCell TransplantationCardiovascular Proceduresmedicine.medical_treatmentCardiomyopathylcsh:Medicine030204 cardiovascular system & hematologyBiochemistryHypoplastic left heart syndromeTissue Culture TechniquesElectrocardiography0302 clinical medicineAnimal CellsHeart RegenerationHypoplastic Left Heart SyndromeNeurobiology of Disease and RegenerationMedicine and Health SciencesMorphogenesisBlood and Lymphatic System ProceduresMyocytes CardiacChildlcsh:ScienceCells CulturedTetralogy of FallotMultidisciplinaryStem CellsStem Cell TherapyDilated cardiomyopathyHeartStem-cell therapyCardiac Transplantationmedicine.anatomical_structureNeurologyChild PreschoolCardiologyTetralogy of Fallotcardiovascular systemStem cellCellular TypesAnatomyResearch ArticleCardiomyopathy Dilatedmedicine.medical_specialtyAdolescentHeart VentriclesSurgical and Invasive Medical Procedures03 medical and health sciencesInternal medicinemedicineHumansRegenerationVimentincardiovascular diseasesClinical GeneticsTransplantationbusiness.industrylcsh:RInfant NewbornCorrectionInfantBiology and Life SciencesProteinsMesenchymal Stem CellsCell BiologyOrgan Transplantationmedicine.diseaseCytoskeletal Proteins030104 developmental biologyVentricleCardiovascular Anatomylcsh:QbusinessOrganism DevelopmentDevelopmental BiologyStem Cell TransplantationPLoS ONE
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Dominant variants in the splicing factor PUF60 cause a recognizable syndrome with intellectual disability, heart defects and short stature

2016

Item does not contain fulltext Verheij syndrome, also called 8q24.3 microdeletion syndrome, is a rare condition characterized by ante- and postnatal growth retardation, microcephaly, vertebral anomalies, joint laxity/dislocation, developmental delay (DD), cardiac and renal defects and dysmorphic features. Recently, PUF60 (Poly-U Binding Splicing Factor 60 kDa), which encodes a component of the spliceosome, has been discussed as the best candidate gene for the Verheij syndrome phenotype, regarding the cardiac and short stature phenotype. To date, only one patient has been reported with a de novo variant in PUF60 that probably affects function (c.505C>T leading to p.(His169Tyr)) associated wi…

0301 basic medicineMaleMESH: Heart Defects Congenital / physiopathologyMicrocephalyPathologyMESH: Heart Defects Congenital / geneticsMESH: Exome / genetics030105 genetics & heredityMESH: RNA Splicing / geneticsMicrophthalmia[SDV.MHEP.MI]Life Sciences [q-bio]/Human health and pathology/Infectious diseasesMESH: ChildExomeMESH: RNA Splicing Factors / geneticsChildFrameshift MutationMESH: High-Throughput Nucleotide SequencingGenetics (clinical)Exome sequencingColobomaMESH: Frameshift MutationHigh-Throughput Nucleotide SequencingMicrodeletion syndromeMicrocephaly Verheij syndrome PUF60ChemistryPhenotypeChild PreschoolDISEASESMicrocephalyMedical geneticsFemaleRNA Splicing Factorsmedicine.symptomChromosome DeletionChromosomes Human Pair 8MESH: Dwarfism / genetics*Heart Defects Congenitalmedicine.medical_specialtyGENESAdolescentRNA SplicingMESH: Chromosome DeletionDwarfismBiologyMESH: PhenotypeShort statureArticlePUF6003 medical and health sciencesInternal medicineIntellectual Disability[ SDV.MHEP ] Life Sciences [q-bio]/Human health and pathologyGeneticsmedicineHumansCraniofacialBiologyMESH: AdolescentNeurodevelopmental disorders Donders Center for Medical Neuroscience [Radboudumc 7]MESH: HumansMESH: Child Preschoolmedicine.diseaseMESH: Repressor Proteins / geneticsMESH: MaleRepressor Proteins030104 developmental biologyEndocrinologyMESH: Chromosomes Human Pair 8 / geneticsMESH: Dwarfism / physiopathologyMESH: Intellectual Disability / physiopathologyHuman medicineMESH: Intellectual Disability / geneticsVerheij syndromeMESH: Female[SDV.MHEP]Life Sciences [q-bio]/Human health and pathology
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A systematic variant screening in familial cases of congenital heart defects demonstrates the usefulness of molecular genetics in this field

2016

International audience; The etiology of congenital heart defect (CHD) combines environmental and genetic factors. So far, there were studies reporting on the screening of a single gene on unselected CHD or on familial cases selected for specific CHD types. Our goal was to systematically screen a proband of familial cases of CHD on a set of genetic tests to evaluate the prevalence of disease-causing variant identification. A systematic screening of GATA4, NKX2-5, ZIC3 and Multiplex ligation-dependent probe amplification (MLPA) P311 Kit was setup on the proband of 154 families with at least two cases of non-syndromic CHD. Additionally, ELN screening was performed on families with supravalvula…

0301 basic medicineProbandMaleCardiomyopathy22q11.2Disease030204 cardiovascular system & hematologyBioinformatics0302 clinical medicinede-novoEpidemiology3 large registriesGenetics (clinical)zic3 mutationsGeneticsHigh-Throughput Nucleotide Sequencing3. Good healthPedigreeHomeobox Protein Nkx-2.5malformationsFemaleepidemiologyHeart Defects Congenitalmedicine.medical_specialtyGenetic counselingArticle03 medical and health sciences[ SDV.MHEP ] Life Sciences [q-bio]/Human health and pathologyMolecular geneticsGeneticsmedicineHumansMultiplex ligation-dependent probe amplificationGenetic TestingHomeodomain Proteinsdiseasebusiness.industryvariabilityGenetic Variationmedicine.diseaseGATA4 Transcription Factor030104 developmental biologyMutationEtiologycardiovascular defectsbusinessMultiplex Polymerase Chain Reactioncardiomyopathy[SDV.MHEP]Life Sciences [q-bio]/Human health and pathologyTranscription Factors
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Susceptibility to Heart Defects in Down Syndrome Is Associated with Single Nucleotide Polymorphisms in HAS 21 Interferon Receptor Cluster and VEGFA G…

2020

Background: Congenital heart defects (CHDs) are present in about 40&ndash

0301 basic medicineVEGFAAdultHeart Defects CongenitalMaleVascular Endothelial Growth Factor ADown syndromelcsh:QH426-470AdolescentChromosomes Human Pair 21Down syndromeSNPSingle-nucleotide polymorphismBiologyPolymorphism Single NucleotideArticle03 medical and health sciencesHeart disorder0302 clinical medicineGenotypeGeneticsmedicineHumansGeneGenetics (clinical)IFNRReceptors InterferonGeneticsmedicine.diseasePhenotypeHeart defectlcsh:GeneticsVascular endothelial growth factor A030104 developmental biologySettore MED/03 - Genetica Medica030220 oncology & carcinogenesisMultigene Familyheart defectsFemaleChromosome 21SNPsGenes
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Effects of innovative and conventional sanitizing treatments on the reduction of Saccharomycopsis fibuligera defects on industrial durum wheat bread

2016

Wickerhamomyces anomalus, Hyphopichia burtonii and Saccharomycopsis fibuligera are spoilage yeasts causing chalk mold defects on sliced bread packaged under modified atmosphere. The first objective of this study, carried out in a bread-making company for two consecutive years, was to genetically identify yeasts isolated from spoiled sliced bread in Modified Atmosphere Packaging (MAP) and to determine the dominant species among identified strains. The second objective was to evaluate the effects of hydrogen peroxide and silver solution 12% (HPS) treatment in the leavening cells and cooling chambers, in comparison with the conventional Ortho-Phenylphenol (OPP) fumigating treatment, on the inc…

0301 basic medicineWater activityBaked goodsWickerhamomyces anomalus030106 microbiologyFood spoilageSaccharomycopsismedicine.disease_causeMicrobiology03 medical and health sciences0404 agricultural biotechnologyYeastsMoldmedicineBaked goodFood scienceChalk mold defects; Baked goods; 26S sequencing; MAP packaging; Water activity; Environmental biodecontaminationTriticumLeavening agent26S sequencingChalk mold defectChemistryEnvironmental biodecontaminationTemperatureMAP packagingWaterBreadHydrogen Peroxide04 agricultural and veterinary sciencesGeneral MedicineSettore AGR/15 - Scienze E Tecnologie AlimentariContamination040401 food scienceYeastRNA RibosomalModified atmosphereChalk mold defectsFood ScienceWater activity
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In Vivo Cardiotoxicity Induced by Sodium Aescinate in Zebrafish Larvae

2016

Sodium aescinate (SA) is a widely-applied triterpene saponin product derived from horse chestnut seeds, possessing vasoactive and organ-protective activities with oral or injection administration in the clinic. To date, no toxicity or adverse events in SA have been reported, by using routine models (in vivo or in vitro), which are insufficient to predict all aspects of its pharmacological and toxicological actions. In this study, taking advantage of transparent zebrafish larvae (Danio rerio), we evaluated cardiovascular toxicity of SA at doses of 1/10 MNLC, 1/3 MNLC, MNLC and LC10 by yolk sac microinjection. The qualitative and quantitative cardiotoxicity in zebrafish was assessed at 48 h p…

0301 basic medicinesodium aescinateEmbryo NonmammalianHeart malformationDrug Evaluation PreclinicalPharmaceutical Science010501 environmental sciencesPharmacology01 natural sciencesAnalytical ChemistryHeart RateDrug DiscoveryToxicity Tests ChronicZebrafishYolk SacbiologyCommunicationHeartLC10medicine.anatomical_structureChemistry (miscellaneous)LarvaToxicityMolecular MedicineHeart Defects CongenitalMicroinjectionscardiotoxicityHemorrhagelarvaelcsh:QD241-44103 medical and health scienceslcsh:Organic chemistryIn vivoHeart ratemedicineMNLCAnimalsPhysical and Theoretical ChemistryYolk sacAdverse effect0105 earth and related environmental sciencesCardiotoxicityDose-Response Relationship DrugOrganic ChemistryThrombosisSaponinsbiology.organism_classificationzebrafishTriterpenes030104 developmental biologyMolecules
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Quando l’amnios si rompe troppo presto e… da solo

2022

Rupture of the amniochorionic membranes is a recognized risk factor in the second and third trimester for chorioamnionitis, sepsis and preterm delivery. The isolated rupture of amnion is conversely responsible for a malformation spectrum known as early amnion rupture sequence or ADAM (Amniotic Deformity, Adhesions, Mutilations) complex, characterized by asymmetrical congenital anomalies, including limb reduction and craniofacial defects, usually arising during the 2nd or 3rd trimester of gestation. The paper reports the case of a 4-month-old infant who was referred for genetic counselling in the suspicion of malformation syndrome with asymmetrical limb reduction defects. The present case un…

ADAM complex Early amnion rupture sequence Limb reduction defects Genetic counselling Multidisciplinary management Diagnosis communicationPediatrics Perinatology and Child Health
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Anorectal malformations and pregnancy-related disorders: a registry-based case-control study in 17 European regions

2013

Objective To identify pregnancy-related risk factors for different manifestations of congenital anorectal malformations (ARMs).Design A population-based case-control study.Setting Seventeen EUROCAT (European Surveillance of Congenital Anomalies) registries, 1980-2008.Population The study population consisted of 1417 cases with ARM, including 648 cases of isolated ARM, 601 cases of ARM with additional congenital anomalies, and 168 cases of ARM-VACTERL (vertebral, anal, cardiac, tracheo-esophageal, renal, and limb defects), along with 13371 controls with recognised syndromes or chromosomal abnormalities.Methods Multiple logistic regression analyses were used to calculate adjusted odds ratios …

ANOMALIESaetiologyMaternalLogistic regressionAnus ImperforateEpilepsyPre-EclampsiaMEDICATION USEPregnancyRisk FactorsOdds RatioAetiologymedia_commoneducation.field_of_studyObstetricsMATERNAL EXPOSUREObstetrics and GynecologyAnal atresiaCONGENITAL-MALFORMATIONSAnorectal MalformationsEuropeParityHuman Reproduction Renal disorder [NCEBP 12]Population studyFemaleAnal Atresiamedicine.medical_specialtyFeverReproductive Techniques Assistedmedia_common.quotation_subjectPopulationFertilityGenomic disorders and inherited multi-system disorders [IGMD 3]VACTERLmedicineanorectal malformations; anal atresia; birth defects; aetiology; maternal; pregnancy; VACTERLHumansAbnormalities MultipleBirth DefectseducationPregnancyEpilepsybusiness.industryRENACCase-control studyInfant NewbornAbortion InducedOdds ratioBIRTH-DEFECTS PREVENTIONBECKWITH-WIEDEMANN-SYNDROMEHuman Reproducion Genomic disorders and inherited multi-system disorders [NCEBP 12]Estados de Saúde e de Doençamedicine.diseasePregnancy Complicationsmaternalbirth defectsEvaluation of complex medical interventions [NCEBP 2]Case-Control StudiesRISK-FACTORSPregnancy TwinMULTIPLE BIRTHSbusinessIN-VITRO FERTILIZATION
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Irradiation effects on the OH-related infrared absorption band in synthetic wet silica.

2007

The effects of b-irradiation on the OH-related infrared (IR) absorption band in synthetic wet silica samples have been investigated by Fourier transform infrared spectroscopy. Depending on the accumulated doses, b-irradiation affects different zones of the IR composite band at about 3670 cm 1 , assigned to the OH stretching modes of silanol groups. These modifications are independent of the original OH content. The results are discussed considering possible radiation-induced changes of the silanol bonding configuration and of the glass network. These are monitored by revealing the IR band a 2260 cm 1 , which is related to the distribution of Si–O–Si bond angle. We have identified the existence of…

Absorption spectroscopyChemistryInfraredPoint defects in silicaAnalytical chemistryInfrared spectroscopyCondensed Matter PhysicsElectronic Optical and Magnetic MaterialsSilanolchemistry.chemical_compoundAbsorption bandMolecular vibrationMaterials ChemistryCeramics and CompositesDynamic properties of vitreous systemsIrradiationFourier transform infrared spectroscopy
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CNGB3 mutations account for 50% of all cases with autosomal recessive achromatopsia

2005

Contains fulltext : 47591.pdf (Publisher’s version ) (Closed access) Achromatopsia is a congenital, autosomal recessively inherited disorder characterized by a lack of color discrimination, low visual acuity (<0.2), photophobia, and nystagmus. Mutations in the genes for CNGA3, CNGB3, and GNAT2 have been associated with this disorder. Here, we analyzed the spectrum and prevalence of CNGB3 gene mutations in a cohort of 341 independent patients with achromatopsia. In 163 patients, CNGB3 mutations could be identified. A total of 105 achromats carried apparent homozygous mutations, 44 were compound (double) heterozygotes, and 14 patients had only a single mutant allele. The derived CNGB3 mutatio…

AchromatopsiaGenetics and epigenetic pathways of disease [NCMLS 6]genetic structuresGATED CATION CHANNELCNGB3 mutationsNonsense mutationMutantCyclic Nucleotide-Gated Cation ChannelsColor Vision DefectsGenes RecessiveLocus (genetics)Gene mutationBiologyTOTAL COLOURBLINDNESSIon ChannelsCLONINGDogscyclic nucleotide-gated channelGNAT2GeneticsmedicineLOCUSAnimalsHumansMissense mutationNeurosensory disorders [UMCN 3.3]ACHM3 locusDog DiseasesAlleleAllelesGenetics (clinical)Geneticstotal colorblindnessGNAT2PHOTORECEPTORSDYSTROPHYmedicine.diseaseCONE DEGENERATIONGENEeye diseasesPhenotypeEvaluation of complex medical interventions [NCEBP 2]MutationRetinal Cone Photoreceptor Cellssense organsachromatopsiarod monochromacyALPHA-SUBUNIThuman activities
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