Search results for "SHP"

showing 10 items of 25 documents

Mutations in SKI in Shprintzen-Goldberg syndrome lead to attenuated TGF-β responses through SKI stabilization.

2020

ABSTRACTShprintzen-Goldberg syndrome (SGS) is a multisystemic connective tissue disorder, with considerable clinical overlap with Marfan and Loeys-Dietz syndromes. These syndromes have commonly been associated with enhanced TGF-β signaling. In SGS patients, heterozygous point mutations have been mapped to the transcriptional corepressor SKI, which is a negative regulator of TGF-β signaling that is rapidly degraded upon ligand stimulation. The molecular consequences of these mutations, however, are not understood. Here we use a combination of structural biology, genome editing and biochemistry to show that SGS mutations in SKI abolish its binding to phosphorylated SMAD2 and SMAD3. This resul…

0301 basic medicineMaleSMADmedicine.disease_causeMarfan SyndromeActivin0302 clinical medicineGenome editingTransforming Growth Factor betaGene expressionBiology (General)MutationShprintzen-Goldberg syndromeGeneral NeuroscienceQRShprintzen–Goldberg syndromeGeneral MedicineLigand (biochemistry)Chromosomes and Gene ExpressionCell biologyDNA-Binding ProteinsMedicinePhosphorylationFemaleSignal TransductionResearch ArticleHumanTGF-βQH301-705.5ScienceBiologyGeneral Biochemistry Genetics and Molecular Biology03 medical and health sciencesCraniosynostosesstomatognathic systemBiochemistry and Chemical BiologyProto-Oncogene ProteinsmedicineHumansGeneral Immunology and MicrobiologyPoint mutationmedicine.diseaseSKIArachnodactyly030104 developmental biologyStructural biologyMutation030217 neurology & neurosurgerySMADTransforming growth factoreLife
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A Novel Solution to Find the Dynamic Response of an Euler–Bernoulli Beam Fitted with Intraspan TMDs under Poisson Type Loading

2020

This contribution considers a virtual experiment on the vibrational response of rail and road bridges equipped with smart devices in the form of damping elements to mitigate vibrations. The internal damping of the bridge is considered a discontinuity that contain a dashpot. Exact complex eigenvalues and eigenfunctions are derived from a characteristic equation built as the determinant of a 4 x 4 matrix

Computer science020101 civil engineeringPoissonian Loading02 engineering and technologylcsh:TechnologyDashpot0201 civil engineeringDamper0203 mechanical engineeringTuned mass damperGeneral Materials ScienceEigenvalues and eigenvectorsCivil and Structural EngineeringGeneralized functionTuned Mass Damperlcsh:TMathematical analysisCharacteristic equationBuilding and ConstructionWhite noiseGeotechnical Engineering and Engineering GeologyComputer Science ApplicationsVibration020303 mechanical engineering & transportsEuler Bernoulli BeamEuler Bernoulli beam Poissonian loading Tuned mass damperSettore ICAR/08 - Scienza Delle CostruzioniInfrastructures
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Aprendiendo Vibraciones Mec´anicas con Wolfram Mathematica

2015

[EN] Mechanical vibrations as subject can be found within many Engineering and Science Degrees. To achieve that the students understand the mathematics and its physical interpretation is the objective we should get as docents. In this paper we describe how to create a simple graphical model of a single degree of freedom vibrating system allowing us to visualize concepts like above concepts damping, resonance or forced vibrations. For that, we use the popular symbolic software Wolfram Mathematica with which, without an excessive programming complexity, we can obtain a very satisfactory visual model capable to move itself, controlled by parameters. In addition, the model incorporates the curv…

Computer scienceVibraciones mecánicasMechanical vibrationsmechanical vibrationsWolfram Mathematicalcsh:Education (General)Animación en el tiempoSoftwareCalculusmass-spring-dashpot systemGraphical modelSimulationInterpretation (logic)Graphical representationbusiness.industryMass-spring-dashpot systemtime domain animationTime domain animationVibrationgraphical representationRepresentación gráficalcsh:L7-991Single degree of freedombusinessSistema masa-muelle-amortiguador
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The influence of addition of Borago officinalis with antibacterial activity on the sensory quality of fresh pasta

2015

Abstract Borage (Borago officinalis L.) is a herbaceous plant of the Boraginaceae family cultivated throughout the world for several purposes, including food preparations, mainly beverages and salads. Some Italian recipes use borage as a food ingredient, in particular as condiment for pasta. The aqueous extract (AE) from borage leaves can act as biopreservative in foods due to its inhibition towards the main foodborne pathogen bacteria. Fresh pasta, due to the high content of water, is a food product with a limited shelf life. In order to test the suitability of borage to produce fresh pasta with a prolonged shelf life, borage AE was used in dried form as a raw material for the production o…

Cultural StudiesPreservativeFresh pastaFlavourSettore AGR/04 - Orticoltura E FloricolturaBorageShelf lifeIngredientFreshpastaFood scienceSensoryevaluationBorageSensory evaluationbiologyAqueous extractsBoraginaceaebiology.organism_classificationHorticultureAqueous extractAntibacterialactivityOfficinalisAqueous extracts;Antibacterialactivity;Borage;Freshpasta;SensoryevaluationBoragoAntibacterial activityFood ScienceSettore AGR/16 - Microbiologia AgrariaInternational Journal of Gastronomy and Food Science
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Molecular and patho-physiological basis of syndromes with developmental anomalies and intellectual disability

2013

Intellectual disability (ID) corresponds to abnormal intellectual performances and adaptive functions, beginning in childhood. It is estimated that 2-3% of individuals develop a ID, which represents a significant medical challenge since people with ID are frequently in situations of social dependence. Overall, a critical involvement of genetic factors in this disease is suspected. To date, several hundreds of genes are known to be responsible for ID. The ID is particularly characterized by extreme clinical and genetic heterogeneity, that made it resistant to conventional genetic studies. However, it is classicaly separated between syndromic ID, which may be clinically recognizable due to as…

Exome sequencingMendelian disorders[SDV.MHEP] Life Sciences [q-bio]/Human health and pathologyShprintzen-Goldberg syndromeIntellectual disabilitySyndromes microdélétionnels[SDV.GEN.GH] Life Sciences [q-bio]/Genetics/Human geneticsAnomalies du développementDéficience intellectuelleSéquençage d’exomeMicrodeletionnal syndromesSyndrome de Shprintzen-Goldberg[SDV.BDD] Life Sciences [q-bio]/Development BiologyMultiple congenital anomalies[SDV.BBM] Life Sciences [q-bio]/Biochemistry Molecular BiologyMaladies mendéliennes
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Exact mechanical models of fractional hereditary materials

2012

Fractional Viscoelasticity is referred to materials, whose constitutive law involves fractional derivatives of order β R such that 0 β 1. In this paper, two mechanical models with stress-strain relation exactly restituting fractional operators, respectively, in ranges 0 β 1 / 2 and 1 / 2 β 1 are presented. It is shown that, in the former case, the mechanical model is described by an ideal indefinite massless viscous fluid resting on a bed of independent springs (Winkler model), while, in the latter case it is a shear-type indefinite cantilever resting on a bed of independent viscous dashpots. The law of variation of all mechanical characteristics is of power-law type, strictly related to th…

Hereditary materialMechanical EngineeringMathematical analysisConstitutive equationFractional derivativeType (model theory)Viscous liquidCondensed Matter PhysicsPower lawViscoelasticityDashpotFractional calculusClassical mechanicsMechanical fractancePower-lawsMechanics of MaterialsGeneral Materials ScienceIdeal (ring theory)Settore ICAR/08 - Scienza Delle CostruzioniFractional integralMathematicsJournal of Rheology
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Multicolor upconversion luminescence of GdVO4:Ln3+/Yb3+ (Ln3+ = Ho3+, Er3+, Tm3+, Ho3+/Er3+/Tm3+) nanorods

2016

Lanthanide-doped GdVO4 nanorods that exhibit upconversion emission under 982 nm excitation have been prepared by a facile room-temperature chemical co-precipitation method followed by a subsequent annealing at temperatures of 600 degrees C, 800 degrees C and 1000 degrees C. Multicolor upconversion emission, including white, was achieved by tuning the concentrations of dopant lanthanide ions (Ho3+, Er3+, Tm3+ and Yb3+) in GdVO4. It is found that four GdVO4 samples emit light with the white chromaticity coordinates of (0.326, 0.339), (0.346, 0.343), (0.323, 0327) and (0.342, 0.340) respectively, under a single-wavelength NIR excitation. These coordinates are very close to the standard equal e…

LanthanideMaterials scienceDopantWhite lightAnnealing (metallurgy)Process Chemistry and TechnologyGeneral Chemical EngineeringMulticolor emissionAnalytical chemistry02 engineering and technologyPhoshpors010402 general chemistry021001 nanoscience & nanotechnology01 natural sciencesPhoton upconversion0104 chemical sciencesIonLanthanidesNanorodVanadatesChromaticity0210 nano-technologyExcitationUpconversionDyes and Pigments
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On the reception of Wilhelm von Humboldt’s linguistic ideas in the Soviet Union from the late 1920s to the early 1950s

2015

The present article discusses the Soviet reception of Humboldt’s linguistic ideas, focusing on different interpretations of his ideas during the period between the latter half of the 1920s and the early 1950s. While Humboldt’s idea of the inner form of language was an important ingredient in Shpet’s phenomenology, the attitude towards Humboldt changed radically in the late 1920s and early 1930s when the ‘bolshevization’ of the sciences had reached linguistics. The idea that language, nation, and culture are closely interconnected was at odds with the ‘Marxist’ idea of class-language, according to which linguistic diversity derives from the socio-economic characteristics of societies. In the…

Linguistics and LanguageHistoryinner formmedia_common.quotation_subjectta6121GustavSoviet scienceLanguage and LinguisticsPhenomenology (philosophy)Politicssymbols.namesakeShpetStalinJosephMarxist philosophyta615Linguistic relativityWilhelm vonSoviet linguisticsmedia_commonHumboldtLinguistic diversityLinguisticsMarrismsymbolsIdeologySoviet unionIazykfront groupClassicsLanguage and History
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In-Frame Mutations in Exon 1 of SKI Cause Dominant Shprintzen-Goldberg Syndrome

2012

International audience; Shprintzen-Goldberg syndrome (SGS) is characterized by severe marfanoid habitus, intellectual disability, camptodactyly, typical facial dysmorphism, and craniosynostosis. Using family-based exome sequencing, we identified a dominantly inherited heterozygous in-frame deletion in exon 1 of SKI. Direct sequencing of SKI further identified one overlapping heterozygous in-frame deletion and ten heterozygous missense mutations affecting recurrent residues in 18 of the 19 individuals screened for SGS; these individuals included one family affected by somatic mosaicism. All mutations were located in a restricted area of exon 1, within the R-SMAD binding domain of SKI. No mut…

MaleModels Molecularmedicine.disease_cause[SDV.BBM.BM] Life Sciences [q-bio]/Biochemistry Molecular Biology/Molecular biologyMarfan SyndromeArachnodactylyExon0302 clinical medicineGene OrderMissense mutationGenetics(clinical)Child[ SDV.GEN.GH ] Life Sciences [q-bio]/Genetics/Human geneticsGenetics (clinical)Exome sequencingGenes DominantGenetics0303 health sciencesMutationShprintzen–Goldberg syndromeExonsPhenotypePedigreeDNA-Binding ProteinsPhenotypeChild PreschoolFemalemedicine.symptomAdultAdolescentMolecular Sequence Data[ SDV.BBM.BM ] Life Sciences [q-bio]/Biochemistry Molecular Biology/Molecular biologyBiology[SDV.GEN.GH] Life Sciences [q-bio]/Genetics/Human genetics03 medical and health sciencesCamptodactylyCraniosynostosesYoung Adultstomatognathic systemReportProto-Oncogene ProteinsmedicineGeneticsHumansAmino Acid Sequence030304 developmental biologyFacies[SDV.BBM.BM]Life Sciences [q-bio]/Biochemistry Molecular Biology/Molecular biologymedicine.diseaseMolecular biologyProtein Structure TertiaryArachnodactyly[SDV.GEN.GH]Life Sciences [q-bio]/Genetics/Human geneticsMutationSequence Alignmenthuman activities030217 neurology & neurosurgery
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Linear and nonlinear fractional hereditary constitutive laws of asphalt mixtures

2016

The aim of this paper is to propose a fractional viscoelastic and viscoplastic model of asphalt mixtures using experimental data of several tests such as creep and creep recovery performed at different temperatures and at different stress levels. From a best fitting procedure it is shown that both the creep one and recovery curve follow a power law model. It is shown that the suitable model for asphalt mixtures is a dashpot and a fractional element arranged in series. The proposed model is also available outside of the linear domain but in this case the parameters of the model depend on the stress level.

Materials scienceasphalt mixtureStrategy and Managementcreep test0211 other engineering and technologies02 engineering and technologyfractional calculusPower lawcreep test.ViscoelasticityDashpot0203 mechanical engineering021105 building & constructionSettore ICAR/04 - Strade Ferrovie Ed AeroportiComposite materialviscoplasticityviscoelasticityCivil and Structural EngineeringBuilding constructionmechanical modelsViscoplasticityMechanicsmechanical modelFractional calculusfractional calculus asphalt mixture viscoelasticity viscoplasticity rheology mechanical models creep testfractional calculuNonlinear system020303 mechanical engineering & transportsCreepAsphaltrheologyTH1-9745
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