Search results for "RARE"

showing 10 items of 2778 documents

Amorphous polyphosphate–hydroxyapatite: A morphogenetically active substrate for bone-related SaOS-2 cells in vitro

2015

There is increasing evidence that inorganic calcium-polyphosphates (polyP) are involved in human bone hydroxyapatite (HA) formation. Here we investigated the morphology of the particles, containing calcium phosphate (CaP) with different concentrations of various Na-polyP concentrations, as well as their effects in cell culture. We used both SaOS-2 cells and human mesenchymal stem cells. The polymeric phosphate readily binds calcium ions under formation of insoluble precipitates. We found that addition of low concentrations of polyP (10wt.%, referred to the CaP deposits) results in an increased size of the HA crystals. Surprisingly, at higher polyP concentrations (10wt.%) the formation of cr…

0301 basic medicinePolymersBiocompatible Materials02 engineering and technologyBone tissueBiochemistryApatitechemistry.chemical_compoundX-Ray DiffractionOsteogenesisPolyphosphatesSpectroscopy Fourier Transform InfraredTissue ScaffoldsBiomaterialGeneral Medicine021001 nanoscience & nanotechnologyMicrospheresGene Expression Regulation Neoplasticmedicine.anatomical_structureBiochemistryvisual_artvisual_art.visual_art_mediumAlkaline phosphataseHydroxyapatites0210 nano-technologyBiotechnologyMaterials scienceBiocompatibilityBiomedical Engineeringchemistry.chemical_elementCalciumCollagen Type IBiomaterials03 medical and health sciencesCalcification PhysiologicMicroscopy Electron TransmissionCell Line TumormedicineHumansBone regenerationMolecular BiologyCell ProliferationIonsOsteoblastsTissue EngineeringSodiumMesenchymal Stem CellsAlkaline PhosphatasePhosphateMicroscopy ElectronDurapatite030104 developmental biologychemistryBiophysicsCalciumActa Biomaterialia
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Next-generation sequencing in clinical practice: from the patients' preferences to the informed consent process

2016

International audience

0301 basic medicineProcess (engineering)Pilot Projects030105 genetics & heredityNext-generation sequencing NGS03 medical and health sciencesRare Diseasespréférences de spatientsNursingInformed consentHumans[ SHS.ECO ] Humanities and Social Sciences/Economies and financessequençage haut-débitComputingMilieux_MISCELLANEOUSIncidental FindingsInformed ConsentPublic Health Environmental and Occupational HealthHigh-Throughput Nucleotide SequencingPatient PreferenceGeneral Medicineinformed consent processFocus Groups[SHS.ECO]Humanities and Social Sciences/Economics and Financeconsentement informéFocus groupPatient preferenceClinical Practicepatients' preferencesPsychology
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Generation of three human iPSC lines from PLAN (PLA2G6-associated neurodegeneration) patients

2021

© 2021 The Authors.

0301 basic medicineQH301-705.5Cellular differentiationInduced Pluripotent Stem CellsNeuroaxonal Dystrophies:Cells::Stem Cells::Adult Stem Cells::Induced Pluripotent Stem Cells [ANATOMY]Biologymedicine.disease_cause:células::células madre::células madre adultas::células madre pluripotentes inducidas [ANATOMÍA]Sistema nerviós - DegeneracióCell LineDermal fibroblastGroup VI Phospholipases A203 medical and health sciencesKruppel-Like Factor 40302 clinical medicineSOX2medicineHumans:enfermedades del sistema nervioso::enfermedades neurodegenerativas [ENFERMEDADES]Biology (General)Induced pluripotent stem cellMutationNeurodegenerationCell DifferentiationCell BiologyGeneral Medicinemedicine.diseaseCellular Reprogramming030104 developmental biologyKLF4:Nervous System Diseases::Neurodegenerative Diseases [DISEASES]MutationCancer researchMalalties raresReprogramming030217 neurology & neurosurgeryGenèticaDevelopmental BiologyStem Cell Research
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Nuclear inclusions of pathogenic ataxin-1 induce oxidative stress and perturb the protein synthesis machinery

2020

Spinocerebellar ataxia type-1 (SCA1) is caused by an abnormally expanded polyglutamine (polyQ) tract in ataxin-1. These expansions are responsible for protein misfolding and self-assembly into intranuclear inclusion bodies (IIBs) that are somehow linked to neuronal death. However, owing to lack of a suitable cellular model, the downstream consequences of IIB formation are yet to be resolved. Here, we describe a nuclear protein aggregation model of pathogenic human ataxin-1 and characterize IIB effects. Using an inducible Sleeping Beauty transposon system, we overexpressed the ATXN1(Q82) gene in human mesenchymal stem cells that are resistant to the early cytotoxic effects caused by the expr…

0301 basic medicineSCA1 Spinocerebellar ataxia type-1Intranuclear Inclusion BodiesClinical BiochemistryMSC mesenchymal stem cellProtein aggregationBiochemistry0302 clinical medicineMutant proteinProtein biosynthesisDE differentially expressed genesNuclear proteinlcsh:QH301-705.5FTIR Fourier-transform infrared spectroscopyAtaxin-1lcsh:R5-920biologyChemistryNuclear ProteinspolyQ polyglutamineRibosomeCell biologySB Sleeping BeautyRibosome ; Polyglutamine ; Ataxin-1 ; Oxidative stress ; Transposon ; Sleeping beauty transposon ; Protein networkSpinocerebellar ataxiaProtein foldingCellular modelFunction and Dysfunction of the Nervous Systemlcsh:Medicine (General)Research PaperiPSC induced pluripotent stem cellAtaxin 1Nerve Tissue ProteinsPPI protein-protein interaction03 medical and health sciencesROS reactive oxygen speciesProtein networkSleeping beauty transposonGSEA Gene Set Enrichment AnalysismedicineHumansNPC neural progenitor cellOrganic Chemistrymedicine.diseaseAFM atomic force microscopyOxidative Stress030104 developmental biologylcsh:Biology (General)IIBs intranuclear inclusion bodiesMS mass spectrometryCardiovascular and Metabolic Diseasesbiology.proteinPolyglutamine030217 neurology & neurosurgery
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SPEN haploinsufficiency causes a neurodevelopmental disorder overlapping proximal 1p36 deletion syndrome with an episignature of X chromosomes in fem…

2021

Contains fulltext : 231702.pdf (Publisher’s version ) (Closed access) Deletion 1p36 (del1p36) syndrome is the most common human disorder resulting from a terminal autosomal deletion. This condition is molecularly and clinically heterogeneous. Deletions involving two non-overlapping regions, known as the distal (telomeric) and proximal (centromeric) critical regions, are sufficient to cause the majority of the recurrent clinical features, although with different facial features and dysmorphisms. SPEN encodes a transcriptional repressor commonly deleted in proximal del1p36 syndrome and is located centromeric to the proximal 1p36 critical region. Here, we used clinical data from 34 individuals…

0301 basic medicineSHARPMaleobesitygenotype-phenotype correlationsAutism Spectrum DisorderPROTEINChromosome DisordersHaploinsufficiencyRNA-Binding ProteinPHENOTYPE CORRELATIONS1p36; distal 1p36 deletion syndrome; DNA methylome analysis; episignature; genotype-phenotype correlations; neurodevelopmental disorder; obesity; proximal 1p36 deletion syndrome; SPEN; X chromosome; Adolescent; Autism Spectrum Disorder; Child; Child Preschool; Chromosome Deletion; Chromosome Disorders; Chromosomes Human Pair 1; Chromosomes Human X; DNA Methylation; DNA-Binding Proteins; Epigenesis Genetic; Female; Haploinsufficiency; Humans; Intellectual Disability; Male; Neurodevelopmental Disorders; Phenotype; RNA-Binding Proteins; Young AdultEpigenesis GeneticX chromosome0302 clinical medicineNeurodevelopmental disorderNeurodevelopmental DisorderIntellectual disabilityMOLECULAR CHARACTERIZATIONdistal 1p36 deletion syndromeChildGenetics (clinical)X chromosomeGeneticsXDNA methylome analysiRNA-Binding ProteinsSPLIT-ENDSHypotoniaDNA-Binding ProteinsPhenotypeAutism spectrum disorderChromosomes Human Pair 1Child PreschoolDNA methylome analysisMONOSOMY 1P36Pair 1SPENFemalemedicine.symptomChromosome DeletionHaploinsufficiencyRare cancers Radboud Institute for Health Sciences [Radboudumc 9]HumanAdolescentDNA-Binding ProteinBiologygenotype-phenotype correlationChromosomes03 medical and health sciencesYoung AdultGeneticSDG 3 - Good Health and Well-beingReportIntellectual DisabilityREVEALSGeneticsmedicineHumansEpigeneticsPreschoolChromosomes Human XNeurodevelopmental disorders Donders Center for Medical Neuroscience [Radboudumc 7]1p361p36 deletion syndromeIDENTIFICATIONMUTATIONSproximal 1p36 deletion syndromeDNA Methylationmedicine.diseaseneurodevelopmental disorderGENEepisignature030104 developmental biologyChromosome DisorderNeurodevelopmental Disorders030217 neurology & neurosurgeryEpigenesis
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Heat- and pH-induced BSA conformational changes, hydrogel formation and application as 3D cell scaffold

2016

Aggregation and gelation of globular proteins can be an advantage to generate new forms of nanoscale biomaterials based on the fibrillar architecture. Here, we report results obtained by exploiting the proteins' natural tendency to self-organize in 3D network, for the production of new material based on BSA for medical application. In particular, at five different pH values the conformational and structural changes of the BSA during all the steps of the thermal aggregation and gelation have been analyzed by FTIR spectroscopy. The macroscopic mechanical properties of these hydrogels have been obtained by rheological measurements. The microscopic structure of the gels have been studied by AFM…

0301 basic medicineScaffoldHot TemperatureNanostructureBSACell SurvivalProtein ConformationGlobular proteinBiophysics?-aggregatesBiocompatible Materials02 engineering and technologymacromolecular substancesMicroscopy Atomic ForceBiochemistryMice03 medical and health sciencesProtein structureRheologySpectroscopy Fourier Transform Infraredβ-aggregateAnimalsCell-scaffoldFourier transform infrared spectroscopyMolecular BiologyNanoscopic scalechemistry.chemical_classificationTissue ScaffoldsChemistrySerum Albumin BovineHydrogelsHydrogen-Ion Concentration021001 nanoscience & nanotechnologySettore FIS/07 - Fisica Applicata(Beni Culturali Ambientali Biol.e Medicin)NanostructuresHydrogelCrystallography030104 developmental biologyMechanical spectraBiophysicChemical engineeringFTIRSelf-healing hydrogelsMicroscopy Electron ScanningCattleStress MechanicalRheology0210 nano-technology
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Microorganism adhesion using silicon dioxide: An experimental study

2020

In this study, spectrophotometry was used to measure changes in the absorbance properties of yeast, Gram-positive, and Gram-negative bacteria after their attachment to silicon dioxide microparticles (silica). The goal of this study was to determine whether spectrophotometry is an effective method to distinguish these microorganisms from one another and determine whether they have an affinity for silicon dioxide. The experiments were performed by examining the light absorption properties of yeast, Gram-positive and Gram-negative bacteria in a spectrophotometer, both with and without silicon dioxide microparticles. The experiments produced a number of promising results. First, the spectrophot…

0301 basic medicineSilicon dioxideMicroorganismMicroorganismsMicrobiologyBiochemistryArticleClinical researchAbsorbance03 medical and health scienceschemistry.chemical_compound0302 clinical medicineSpectrophotometrySilicon dioxidemedicinelcsh:Social sciences (General)Bacterial typeslcsh:Science (General)Infectious diseaseMultidisciplinaryChromatographyBacteriamedicine.diagnostic_testbiologyChemistryNear-infrared spectroscopyLaboratory medicinebiology.organism_classificationYeastYeast030104 developmental biologySpectrophotometrylcsh:H1-99030217 neurology & neurosurgeryBacterialcsh:Q1-390Heliyon
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Pressure effects on α-synuclein amyloid fibrils: An experimental investigation on their dissociation and reversible nature

2017

α–synuclein amyloid fibrils are found in surviving neurons of Parkinson's disease affected patients, but the role they play in the disease development is still under debate. A growing number of evidences points to soluble oligomers as the major cytotoxic species, while insoluble fibrillar aggregates could even play a protection role. In this work, we investigate α–synuclein fibrils dissociation induced at high pressure by means of Small Angle X-ray Scattering and Fourier Transform Infrared Spectroscopy. Fibrils were produced from wild type α–synuclein and two familial mutants, A30P and A53T. Our results enlighten the different reversible nature of α–synuclein fibrils fragmentati…

0301 basic medicineSmall AngleAmyloidHigh-pressureMutantBiophysicsmacromolecular substances010402 general chemistryFibril01 natural sciencesBiochemistryDissociation (chemistry)Scattering03 medical and health scienceschemistry.chemical_compoundX-Ray DiffractionScattering Small AngleSpectroscopy Fourier Transform InfraredPressureHumansPoint MutationFourier transform infrared spectroscopyMolecular BiologySpectroscopyAlpha-synucleinAmyloid; FTIR; High-pressure; SAXS; α-synuclein; Amyloid; Humans; Parkinson Disease; Point Mutation; Pressure; Scattering Small Angle; Solubility; Spectroscopy Fourier Transform Infrared; X-Ray Diffraction; alpha-Synuclein; Biophysics; Biochemistry; Molecular BiologySmall-angle X-ray scatteringWild typeα-synucleinParkinson DiseaseSAXSAmyloid fibril0104 chemical sciences?-synucleinCrystallography030104 developmental biologyBiophysicchemistryFTIRSolubilityFourier Transform InfraredBiophysicsalpha-SynucleinHuman
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Extraordinary stability of hemocyanins from L. polyphemus and E. californicum studied using infrared spectroscopy from 294 to 20 K

2016

International audience; Hemocyanins are large oligomeric respiratory proteins found in many arthropods and molluscs. Here we give infrared spectroscopic evidence of a high stability towards exposure to sub-zero temperatures for hemocyanins from the arthropods Limulus polyphemus and Eurypelma californicum at different pH values. Small but distinct temperature induced changes of the secondary structure were observed, but a stable core of at least 40% α-helical structure is preserved as identified in the infrared spectra obtained between 294 and 20 K. The structural changes differ in detail somewhat for the two hemocyanins, with overall fewer changes observed in the case of E. californicum. No…

0301 basic medicineSpectrophotometry InfraredÉlectrochimieInfraredAnalytical chemistryGeneral Physics and AstronomyInfrared spectroscopySpectroscopieCold treatmentProtein Structure SecondaryArthropod Proteins03 medical and health scienceschemistry.chemical_compoundHorseshoe CrabsAnimalsPhysical and Theoretical ChemistryProtein secondary structurebiologySpectroélectrochimieSpidersbiology.organism_classificationTemperature inducedChimie Physique[CHIM.THEO]Chemical Sciences/Theoretical and/or physical chemistryCrystallography030104 developmental biologyMyoglobinchemistryPolyphemusLimulusHemocyaninsPhysical Chemistry Chemical Physics
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Bioprotection Can Be Tuned with Proper Protein/Saccharide Ratio: The Case of Solid Amorphous Matrices

2018

Saccharides, and in particular trehalose, are well known for their high efficiency in protecting biostructures against adverse environmental conditions. The protein dynamics is known to be highly inhibited in a low-water trehalose host medium, the inhibition being markedly dependent on the amount of residual water. Besides hydration, the protein/sugar ratio is expected to affect the properties of saccharide amorphous matrices. In this work, we report an infrared spectroscopy study in dry amorphous matrices of various sugars (the disaccharides trehalose, maltose, sucrose, and lactose, and the trisaccharide raffinose) containing myoglobin, at different protein/sugar ratios. We analyze the str…

0301 basic medicineSucrose010402 general chemistry01 natural sciences03 medical and health scienceschemistry.chemical_compoundSpectroscopy Fourier Transform InfraredMaterials ChemistryTrisaccharidePhysical and Theoretical ChemistryRaffinoseTrehalose carboxymyoglobin infrared spectroscopySugarchemistry.chemical_classificationCarbon MonoxideMolecular StructureMyoglobinWaterMaltoseTrehaloseSettore FIS/07 - Fisica Applicata(Beni Culturali Ambientali Biol.e Medicin)0104 chemical sciencesSurfaces Coatings and FilmsAmorphous solid030104 developmental biologychemistryMyoglobinChemical engineeringSugars
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