Search results for "Cases"

showing 10 items of 124 documents

Pathogenic DDX3X Mutations Impair RNA Metabolism and Neurogenesis during Fetal Cortical Development.

2020

Summary De novo germline mutations in the RNA helicase DDX3X account for 1%–3% of unexplained intellectual disability (ID) cases in females and are associated with autism, brain malformations, and epilepsy. Yet, the developmental and molecular mechanisms by which DDX3X mutations impair brain function are unknown. Here, we use human and mouse genetics and cell biological and biochemical approaches to elucidate mechanisms by which pathogenic DDX3X variants disrupt brain development. We report the largest clinical cohort to date with DDX3X mutations (n = 107), demonstrating a striking correlation between recurrent dominant missense mutations, polymicrogyria, and the most severe clinical outcom…

0301 basic medicineMaleNeurogenesisMutation MissenseBiologyPathogenesisDEAD-box RNA Helicases03 medical and health sciencesMice0302 clinical medicineGermline mutationStress granuleCell Line TumorPolymicrogyriamedicineMissense mutationAnimalsHumansCells CulturedGeneticsCerebral CortexGeneral NeuroscienceNeurogenesismedicine.diseaseRNA Helicase AMice Inbred C57BL030104 developmental biologyNeurodevelopmental DisordersRNAFemaleDDX3X030217 neurology & neurosurgeryNeuron
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Insights into the inhibited form of the redox-sensitive SufE-like sulfur acceptor CsdE

2017

17 p.-8 fig.

0301 basic medicineProtein ConformationDimerlcsh:MedicineMolecular DynamicsCrystallography X-RayPhysical ChemistryBiochemistryDEAD-box RNA HelicasesMolecular dynamicschemistry.chemical_compoundComputational ChemistryNucleophileBiochemical Simulationslcsh:ScienceMultidisciplinaryCrystallographyChemistryOrganic CompoundsPhysicsEscherichia coli ProteinsCondensed Matter Physics3. Good healthPhysical sciencesChemistryCarbon-Sulfur LyasesBiochemistryCrystal StructureResearch ArticleChemical ElementsProtein subunitChemical physicschemistry.chemical_elementOxidative phosphorylationMolecular Dynamics Simulation03 medical and health sciencesThiolsEscherichia coliSolid State PhysicsProtein Interaction Domains and MotifsChemical BondingOrganic Chemistrylcsh:RChemical CompoundsBiology and Life SciencesComputational BiologyDimers (Chemical physics)Hydrogen BondingCell BiologySulfurAcceptorRedox sensitiveOxidative Stress030104 developmental biologyBiophysicslcsh:QProtein MultimerizationSulfur
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DICER- and MMSET-catalyzed H4K20me2 recruits the nucleotide excision repair factor XPA to DNA damage sites

2017

The endoribonuclease DICER facilitates chromatin decondensation during lesion recognition following UV exposure. Chitale and Richly show that DICER mediates the recruitment of the methyltransferase MMSET, which catalyzes the dimethylation of histone H4 at lysine 20 and facilitates the recruitment of the nucleotide excision repair factor XPA.

0301 basic medicineRibonuclease IIIDNA RepairDNA damageDNA repairUltraviolet Raysgenetic processes27Article24DEAD-box RNA HelicasesHistones03 medical and health sciencesCell Line TumorHumansResearch ArticlesbiologyLysinefungiEndoribonuclease Dicerfood and beverages37Cell BiologyDNA Repair PathwayHistone-Lysine N-MethyltransferaseCell biologyChromatinXeroderma Pigmentosum Group A ProteinRepressor Proteinsenzymes and coenzymes (carbohydrates)030104 developmental biologyHistoneHEK293 Cellsbiology.proteinBiocatalysisDicerNucleotide excision repairDNA DamageThe Journal of Cell Biology
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Evaluation of DNA Methylation Episignatures for Diagnosis and Phenotype Correlations in 42 Mendelian Neurodevelopmental Disorders

2020

Contains fulltext : 218274.pdf (Publisher’s version ) (Closed access) Genetic syndromes frequently present with overlapping clinical features and inconclusive or ambiguous genetic findings which can confound accurate diagnosis and clinical management. An expanding number of genetic syndromes have been shown to have unique genomic DNA methylation patterns (called "episignatures"). Peripheral blood episignatures can be used for diagnostic testing as well as for the interpretation of ambiguous genetic test results. We present here an approach to episignature mapping in 42 genetic syndromes, which has allowed the identification of 34 robust disease-specific episignatures. We examine emerging pa…

0301 basic medicine[SDV]Life Sciences [q-bio]Computational biology030105 genetics & heredityBiologyPediatricsArticleCohort Studiesmolecular diagnostics03 medical and health sciencessymbols.namesakeGenetic HeterogeneityGene duplicationGeneticsHumansHunter-McAlpine syndromeGenetics (clinical)Mass screening030304 developmental biologyEpiSignGenetics0303 health sciencesNeurodevelopmental disorders Donders Center for Medical Neuroscience [Radboudumc 7]DNA methylationGenetic heterogeneity030305 genetics & heredityCorrectionSyndromeDNA MethylationMolecular diagnosticsPhenotypePenetranceHuman genetics3. Good healthepisignaturegenomic DNA030104 developmental biologyPhenotypeNeurodevelopmental DisordersDNA methylationuncertain clinical casesMendelian inheritancesymbolsIdentification (biology)VUS classification
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Lack of evidence of mimivirus replication in human PBMCs

2018

The Acanthamoeba polyphaga mimivirus (APMV) was first isolated during a pneumonia outbreak in Bradford, England, and since its discovery many research groups devoted efforts to understand whether this virus could be associated to human diseases, in particular clinical signs and symptoms of pneumonia. In 2013, we observed cytopathic effect in amoebas (rounding and lysis) inoculated with APMV inoculated PBMCs (peripheral blood mononuclear cell) extracts, and at that point we interpreted those results as mimivirus replication in human PBMCs. Based on these results we decided to further investigate APMV replication in human PBMCs, by transmission electron microscopy (TEM) and qPCR. No viral fac…

0301 basic medicinevirukset030106 microbiologyImmunologymimivirusReal-Time Polymerase Chain ReactionVirus ReplicationMicrobiologyPeripheral blood mononuclear cellVirus03 medical and health sciencesMultiplicity of infectionBacterial ProteinsMicroscopy Electron TransmissionacanthamoebaViral factoryHumansCytopathic effectMimivirusbiologyDNA Helicasesta1182biology.organism_classificationVirologyHelicase GeneAcanthamoeba030104 developmental biologyInfectious DiseasesDNA ViralPBMCsLeukocytes MononuclearMimiviridaeMicrobes and Infection
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Discriminación indirecta por pertenencia a minoría nacional : denegación de prestación de viudedad en el caso de matrimonio celebrado según el rito g…

2021

The commented sentence rejects that the Muñoz Díaz doctrine is applicable to all cases of gypsy marriage. In addition, it considers that the denial of effects to the union celebrated according to said rite is not discriminatory. This conclusion is discussed, understanding that the analysis of the singularities of the gypsy people must lead to the conclusion of the existence of indirect discrimination.

:CIENCIAS JURÍDICAS [UNESCO]Indirect discriminationviudedadgypsy rite 1032 1051understanding that the analysis of the singularities of the gypsy people must lead to the conclusion of the existence of indirect discrimination. Discriminación indirectaminoría nacionalrito gitano2070-8157 22082 Revista Boliviana de Derecho 584568 2021 32 8055248 Discriminación indirecta por pertenencia a minoría nacional denegación de prestación de viudedad en el caso de matrimonio celebrado según el rito gitano. Comentario de la STC de España 1/2021UNESCO::CIENCIAS JURÍDICASAntonio The commented sentence rejects that the Muñoz Díaz doctrine is applicable to all cases of gypsy marriage. In additionwidowhoodde 25 de enero) Folgoso Olmoit considers that the denial of effects to the union celebrated according to said rite is not discriminatory. This conclusion is discussednational minority
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Determinación de la edad de un extranjero indocumentado : A propósito de la sentencia de la audiencia provincial de barcelona, de 24 de enero de 2020

2021

In recent years, immigration of minors has increased, which means that administrations establish a differentiated treatment for this sector of immigration due to the fact that they are minors; There are many aspects to take into account in these cases, including verifying the minority age, a fact that is more complicated when it comes to adolescents. This is what is analyzed by the Judgment of the Provincial Court of Barcelona, of January 24, 2020, which confirms the Resolution of the General Directorate of Attention to Children and Adolescents, which agreed to the closure of the homelessness file and the admission of the plaintiff in a Protection Center until the verification of their pers…

:CIENCIAS JURÍDICAS [UNESCO]de 24 de enero de 2020 Ortega Giménezwhich confirms the Resolution of the General Directorate of Attention to Children and AdolescentsLerdys Saray In recent yearsimmigration of minors has increasedminorityimmigration. 550 575There are many aspects to take into account in these casesincluding verifying the minority ageAlfonsowhich agreed to the closure of the homelessness file and the admission of the plaintiff in a Protection Center until the verification of their personal circumstancesmayoría de edadminoría de edadof January 242020since it did not provide any type of documentation. Menor extranjero no acompañadoinmigraciónHeredia SánchezUnaccompanied foreign minora fact that is more complicated when it comes to adolescents. This is what is analyzed by the Judgment of the Provincial Court of BarcelonaUNESCO::CIENCIAS JURÍDICASwhich means that administrations establish a differentiated treatment for this sector of immigration due to the fact that they are minors2070-8157 22082 Revista Boliviana de Derecho 565487 2021 31 7730068 Determinación de la edad de un extranjero indocumentado A propósito de la sentencia de la audiencia provincial de barcelonamajority
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Italian assisted negotiation: an additional tool to settle matrimonial property regime cases

2021

The assisted negotiation procedure highlighted the need to distinguish the various forms of justice in view to obtaining decisions that can effectively satisfy the interests of the family. In fact the agreements who discipline the family conflicts attributes a central role to the will of the couple in the collaborative search for suitable solutions for the management of the crisis of the emotional bond.

:CIENCIAS JURÍDICAS [UNESCO]métodos alternativos de solución de conflictos. 164 179Negociación asistidaUNESCO::CIENCIAS JURÍDICASprenuptial agreementsfamily conflictsconflictos familiaresManuela The assisted negotiation procedure highlighted the need to distinguish the various forms of justice in view to obtaining decisions that can effectively satisfy the interests of the family. In fact the agreements who discipline the family conflicts attributes a central role to the will of the couple in the collaborative search for suitable solutions for the management of the crisis of the emotional bond. Assisted negotiationalternative dispute resolution.2386-4567 22661 Actualidad jurídica iberoamericana 587897 2021 15 8113555 Italian assisted negotiation an additional tool to settle matrimonial property regime cases Giobbiacuerdos prematrimoniales
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La sentencia y su ejecución en el proyecto dominicano de ley sobre control judicial de la administración pública

2021

The regime of the execution of the sentence is analyzed, starting from the powers of declarative and executive substitution of the contentious-administrative judge. It begins with the reference to the constitutional guidelines of the administrative process. Then, the different modalities of judicial substitution are described in the types of sentences (declarative, constitutive and conviction) regulated in the project, to then refer to the regime of executive seizure of public goods. It ends by describing: the powers granted to the judge to agree indirect measures that assist with the execution of the judgment, the cases of impossibility of execution and the extension of the sentence to thi…

:CIENCIAS JURÍDICAS [UNESCO]the cases of impossibility of execution and the extension of the sentence to third parties. SentenciaMiguel Ángel The regime of the execution of the sentence is analyzedSentenceRepública Dominicanaconstitutive and conviction) regulated in the projectDominican Republic 586 617contentious-administrativejudicial executionUNESCO::CIENCIAS JURÍDICASthe powers granted to the judge to agree indirect measures that assist with the execution of the judgment [to then refer to the regime of executive seizure of public goods. It ends by describing]contencioso-administrativo2070-8157 22082 Revista Boliviana de Derecho 584568 2021 32 8055232 La sentencia y su ejecución en el proyecto dominicano de ley sobre control judicial de la administración pública Torrealba Sánchezejecución judicialstarting from the powers of declarative and executive substitution of the contentious-administrative judge. It begins with the reference to the constitutional guidelines of the administrative process. Thento then refer to the regime of executive seizure of public goods. It ends by describing: the powers granted to the judge to agree indirect measures that assist with the execution of the judgmentthe different modalities of judicial substitution are described in the types of sentences (declarative
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Customised next-generation sequencing multigene panel to screen a large cohort of individuals with chromatin-related disorder

2020

BackgroundThe regulation of the chromatin state by epigenetic mechanisms plays a central role in gene expression, cell function, and maintenance of cell identity. Hereditary disorders of chromatin regulation are a group of conditions caused by abnormalities of the various components of the epigenetic machinery, namely writers, erasers, readers, and chromatin remodelers. Although neurological dysfunction is almost ubiquitous in these disorders, the constellation of additional features characterizing many of these genes and the emerging clinical overlap among them indicate the existence of a community of syndromes. The introduction of high-throughput next generation sequencing (NGS) methods f…

Adenosine TriphosphataseAdultMaleCCCTC-Binding FactorTranscription FactorDNA-Binding Proteinchromatin disorderComputational biologyBiologyDNA HelicaseDNA sequencingEpigenesis GeneticMendelian chromatin disordersLocus heterogeneityDe Lange SyndromeGeneticsmedicineCoffin-Lowry SyndromeHumansGenetic Predisposition to DiseaseEpigeneticsGenetic TestingChildGeneGenetics (clinical)Adenosine Triphosphatasesnext generation sequencingepigeneticsGenetic heterogeneityDNA HelicasesMendelian chromatin disorderHistone-Lysine N-Methyltransferasemedicine.diseaseChromatinChromatinDNA-Binding ProteinsMendelian chromatin disorders; epigenetics; next generation sequencingCohortMutationRelated disorderFemaleMyeloid-Lymphoid Leukemia ProteinepigeneticTranscription FactorsHuman
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