Search results for "IPT"

showing 10 items of 6114 documents

C2orf69 mutations disrupt mitochondrial function and cause a multisystem human disorder with recurring autoinflammation

2021

BACKGROUND. Deciphering the function of the many genes previously classified as uncharacterized open reading frame (ORF) would complete our understanding of a cell’s function and its pathophysiology. METHODS. Whole-exome sequencing, yeast 2-hybrid and transcriptome analyses, and molecular characterization were performed in this study to uncover the function of the C2orf69 gene. RESULTS. We identified loss-of-function mutations in the uncharacterized C2orf69 gene in 8 individuals with brain abnormalities involving hypomyelination and microcephaly, liver dysfunction, and recurrent autoinflammation. C2orf69 contains an N-terminal signal peptide that is required and sufficient for mitochondrial…

0301 basic medicineMicrocephalyRespiratory chainBiologyMitochondrionCell LineMitochondrial ProteinsTranscriptomeMiceOpen Reading Frames03 medical and health sciencesAll institutes and research themes of the Radboud University Medical Center0302 clinical medicineLoss of Function MutationGlycogen branching enzymemedicineAnimalsHumansGeneMice KnockoutGeneticsMetabolic Disorders Radboud Institute for Molecular Life Sciences [Radboudumc 6]Glycogen Debranching Enzyme SystemGeneral Medicinemedicine.diseaseMitochondriaOpen reading frameRenal disorders Radboud Institute for Molecular Life Sciences [Radboudumc 11]030104 developmental biology030220 oncology & carcinogenesisMicrocephalybiology.proteinClinical MedicineSignal transductionGlycogenJournal of Clinical Investigation
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The broad phenotypic spectrum of PPP2R1A -related neurodevelopmental disorders correlates with the degree of biochemical dysfunction

2021

PURPOSE: Neurodevelopmental disorders (NDD) caused by protein phosphatase 2A (PP2A) dysfunction have mainly been associated with de novo variants in PPP2R5D and PPP2CA, and more rarely in PPP2R1A. Here, we aimed to better understand the latter by characterizing 30 individuals with de novo and often recurrent variants in this PP2A scaffolding Aα subunit. METHODS: Most cases were identified through routine clinical diagnostics. Variants were biochemically characterized for phosphatase activity and interaction with other PP2A subunits. RESULTS: We describe 30 individuals with 16 different variants in PPP2R1A, 21 of whom had variants not previously reported. The severity of developmental delay …

0301 basic medicineMicrocephaly[SDV]Life Sciences [q-bio]Intellectual disability030105 genetics & heredityBioinformaticsEpilepsyNeurodevelopmental disorderIntellectual disabilityCOREProtein Phosphatase 2SPECIFICITYGenetics (clinical)PROTEIN PHOSPHATASE 2APhenotypeHypotoniaFAMILY3. Good healthPP2A[SDV] Life Sciences [q-bio]PPP2R1APPP2R5DINSIGHTSintellectual disabilityMicrocephalyMuscle Hypotoniamedicine.symptomLanguage delay[SDV.GEN.GH] Life Sciences [q-bio]/Genetics/Human geneticsArticle03 medical and health sciencesNeurodevelopmental disorder[SDV.BBM] Life Sciences [q-bio]/Biochemistry Molecular BiologymedicineHumans[SDV.BBM]Life Sciences [q-bio]/Biochemistry Molecular BiologyEpilepsybusiness.industryMacrocephalyDEPHOSPHORYLATIONmedicine.diseaseneurodevelopmental disorder030104 developmental biology[SDV.GEN.GH]Life Sciences [q-bio]/Genetics/Human geneticsNeurodevelopmental DisordersSUBUNITepilepsyHuman medicineTAUbusinessTranscription Factors
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Triptans and CGRP blockade - impact on the cranial vasculature.

2017

Abstract The trigeminovascular system plays a key role in the pathophysiology of migraine. The activation of the trigeminovascular system causes release of various neurotransmitters and neuropeptides, including serotonin and calcitonin gene-related peptide (CGRP), which modulate pain transmission and vascular tone. Thirty years after discovery of agonists for serotonin 5-HT1B and 5-HT1D receptors (triptans) and less than fifteen after the proof of concept of the gepant class of CGRP receptor antagonists, we are still a long way from understanding their precise site and mode of action in migraine. The effect on cranial vasculature is relevant, because all specific anti-migraine drugs and mig…

0301 basic medicineMigraine DisordersCalcitonin gene related peptide – CGRPNeuropeptidelcsh:MedicineMigraine modelsReviewTriptansReview ArticleCalcitonin gene-related peptide03 medical and health sciences0302 clinical medicineJournal ArticlemedicineHumansMigraine treatmentReceptorbusiness.industryTriptans Calcitonin gene related peptide – CGRP Anti-CGRP (receptor) monoclonal antibodies – mAbs Middle meningeal artery Middle cerebral arteries Migraine models Magnetic resonance angiography (MRA)Anti-CGRP (receptor) monoclonal antibodies – mAbsTrigeminovascular systemlcsh:RTriptansGeneral MedicineMiddle meningeal arterymedicine.diseaseTryptamines3. Good healthMagnetic resonance angiography (MRA)Middle cerebral arteries030104 developmental biologyAnesthesiology and Pain MedicineMigraineAnesthesiaNeurology (clinical)SerotoninbusinessNeuroscience030217 neurology & neurosurgerymedicine.drugReceptors Calcitonin Gene-Related PeptideThe journal of headache and pain
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Relationship between internal root resorption and dens in dente

2020

Background The aim is to report the treatment and follow-up of three lateral incisors with internal root resorption and dens in dente as a possible cause for their development, managed by root canal treatment and apical obturation with MTA or gutta-percha. Case description This case report presents three clinical cases in which dens invaginatus type 2 is shown as a potential cause for the development of internal root resorption. Two cases were filled with a MTA apical plug technique and one with gutta-percha, and all were follow-up through time. Practical implications The incidence of the association of internal root resorption with dens invaginatus may be underestimated and should be studi…

0301 basic medicineMineral trioxide aggregateRoot canaleducationOdontologíaCase ReportRoot resorptionOperative Dentistry and EndodonticsApical plug03 medical and health sciencesDens invaginatus0302 clinical medicineCirugíamedicineTecnología médicaGeneral DentistryPractical implicationsOrthodonticsbusiness.industry030206 dentistryCase descriptionmedicine.disease:CIENCIAS MÉDICAS [UNESCO]030104 developmental biologymedicine.anatomical_structureUNESCO::CIENCIAS MÉDICASbusinessJournal of Clinical and Experimental Dentistry
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The mitochondrial antioxidant SS-31 increases SIRT1 levels and ameliorates inflammation, oxidative stress and leukocyte-endothelium interactions in t…

2018

AbstractThere is growing focus on mitochondrial impairment and cardiovascular diseases (CVD) in type 2 diabetes (T2D), and the development of novel therapeutic strategies in this context. It is unknown whether mitochondrial-targeting antioxidants such as SS-31 protect sufficiently against oxidative damage in diabetes. We aimed to evaluate if SS-31 modulates SIRT1 levels and ameliorates leukocyte-endothelium interactions, oxidative stress and inflammation in T2D patients. Anthropometric and metabolic parameters were studied in 51 T2D patients and 57 controls. Production of mitochondrial reactive oxygen species (ROS), mitochondrial membrane potential, glutathione content, leukocyte-endotheliu…

0301 basic medicineMitochondrial ROSMaleAntioxidantendocrine system diseasesmedicine.medical_treatmentMitochondrionPharmacologymedicine.disease_causeAntioxidantsLeukocyte-endothelial Interactionschemistry.chemical_compoundSirtuin 1Leukocyteschemistry.chemical_classificationMembrane Potential MitochondrialMultidisciplinaryQRMiddle AgedMitochondriaUp-RegulationMedicineFemalemedicine.symptomOligopeptidesRolling FluxScienceInflammationContext (language use)SIRT1 LevelsArticle03 medical and health sciencesmedicineCell AdhesionHuman Umbilical Vein Endothelial CellsHumansAgedInflammationReactive oxygen speciesTranscription Factor RelAGlutathioneSirtuins (SIRT1)Oxidative Stress030104 developmental biologychemistryDiabetes Mellitus Type 2Case-Control StudiesReactive Oxygen SpeciesLeukocyte Rolling VelocityOxidative stressScientific Reports
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The mitochondria-targeted antioxidant MitoQ modulates oxidative stress, inflammation and leukocyte-endothelium interactions in leukocytes isolated fr…

2016

It is not known if the mitochondria-targeted antioxidants such as mitoquinone (MitoQ) can modulate oxidative stress and leukocyte-endothelium interactions in T2D patients. We aimed to evaluate the beneficial effect of MitoQ on oxidative stress parameters and leukocyte-endothelium interactions in leukocytes of T2D patients. The study population consisted of 98 T2D patients and 71 control subjects. We assessed metabolic and anthropometric parameters, mitochondrial reactive oxygen species (ROS) production, glutathione peroxidase 1 (GPX-1), NFκB-p65, TNFα and leukocyte-endothelium interactions. Diabetic patients exhibited higher weight, BMI, waist circumference, SBP, DBP, glucose, insulin, HOMA…

0301 basic medicineMitochondrial ROSMaleGPX1Antioxidantendocrine system diseasesUbiquinonemedicine.medical_treatmentBMI body mass indexClinical BiochemistryLDL low density lipoprotein cholesterolAnti-Inflammatory AgentsTPP triphenylphosphonium030204 cardiovascular system & hematologyPharmacologymedicine.disease_causeBiochemistryMitoQAntioxidantschemistry.chemical_compound0302 clinical medicineGlutathione Peroxidase GPX1IR insulin resistanceLeukocyteslcsh:QH301-705.5chemistry.chemical_classificationlcsh:R5-920AnthropometryChemistryGlutathione peroxidaseType 2 diabetesMiddle AgedFemalemedicine.symptomlcsh:Medicine (General)Research PaperPMN polymorphonuclear leukocyteshs-CRP high-sensitive C-reactive proteinHOMA-IR homeostasis model assessment of insulin resistanceInflammationT2D type 2 diabetes03 medical and health sciencesOrganophosphorus CompoundsmedicineDBP diastolic blood pressure HbA1c glycated hemoglobinHUVEC human umbilical vein endothelial cellsHumansEndotheliumAgedInflammationReactive oxygen speciesMitoQGlutathione PeroxidaseTumor Necrosis Factor-alphaSBP systolic blood pressureOrganic ChemistryTranscription Factor RelAnutritional and metabolic diseasesHDL high density lipoprotein cholesterolOxidative Stress030104 developmental biologylcsh:Biology (General)Diabetes Mellitus Type 2ImmunologyReactive Oxygen SpeciesOxidative stressRedox Biology
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Ribosome-Targeting Antibiotics Impair T Cell Effector Function and Ameliorate Autoimmunity by Blocking Mitochondrial Protein Synthesis

2019

Summary While antibiotics are intended to specifically target bacteria, most are known to affect host cell physiology. In addition, some antibiotic classes are reported as immunosuppressive for reasons that remain unclear. Here, we show that Linezolid, a ribosomal-targeting antibiotic (RAbo), effectively blocked the course of a T cell-mediated autoimmune disease. Linezolid and other RAbos were strong inhibitors of T helper-17 cell effector function in vitro, showing that this effect was independent of their antibiotic activity. Perturbing mitochondrial translation in differentiating T cells, either with RAbos or through the inhibition of mitochondrial elongation factor G1 (mEF-G1) progressi…

0301 basic medicineMitochondrial translationmedicine.medical_treatmentT-LymphocytesCellMitochondrionmedicine.disease_causeRibosomemitochondrial translationOxidative PhosphorylationantibioticsAutoimmunityACTIVATIONMice0302 clinical medicineribosome-targetingMedicine and Health SciencesImmunology and AllergyTRANSCRIPTION FACTORMolecular Targeted TherapyMice Knockout0303 health sciencesEffectorExperimental autoimmune encephalomyelitisautoimmunityCell DifferentiationPeptide Elongation Factor GAnti-Bacterial Agents3. Good healthCell biologymitochondriaInfectious DiseasesCytokinemedicine.anatomical_structureRESPIRATION030220 oncology & carcinogenesisEncephalomyelitis Autoimmune ExperimentalMultiple SclerosisT cellImmunologyINHIBITIONT cellsBiologyOXAZOLIDINONEPeptides CyclicArticleMitochondrial Proteins03 medical and health sciencesNAD+medicineAnimalsHumanselongation factor G1030304 developmental biologyAutoimmune diseaseBacteriaLinezolidBiology and Life SciencesPATHWAYSDNANADmedicine.diseaseIn vitroMice Inbred C57BL030104 developmental biologyTh17 CellsArgyrinCHLORAMPHENICOLMEMBRANERibosomesImmunity
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Conformational dynamism for DNA interaction in the Salmonella RcsB response regulator

2017

17 páginas, 7 figuras, 1 tabla

0301 basic medicineModels MolecularSalmonella typhimuriumProtein Data Bank (RCSB PDB)Plasma protein bindingBiologyCrystallography X-RayDNA-binding protein03 medical and health sciencesBacterial ProteinsProtein DomainsStructural BiologyGeneticsAmino Acid SequencePhosphorylationTranscription factorSequence Homology Amino AcidEffectorPromoterDNACell biologyResponse regulator030104 developmental biologyRegulonBiochemistryNucleic Acid ConformationProtein BindingNucleic Acids Research
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Investigation on Quantitative Structure-Activity Relationships of 1,3,4-Oxadiazole Derivatives as Potential Telomerase Inhibitors.

2020

Background:Telomerase, a reverse transcriptase, maintains telomere and chromosomes integrity of dividing cells, while it is inactivated in most somatic cells. In tumor cells, telomerase is highly activated, and works in order to maintain the length of telomeres causing immortality, hence it could be considered as a potential marker to tumorigenesis.A series of 1,3,4-oxadiazole derivatives showed significant broad-spectrum anticancer activity against different cell lines, and demonstrated telomerase inhibition.Methods:This series of 24 N-benzylidene-2-((5-(pyridine-4-yl)-1,3,4-oxadiazol-2yl)thio)acetohydrazide derivatives as telomerase inhibitors has been considered to carry out QSAR studies…

0301 basic medicineModels MolecularTelomeraseQuantitative structure–activity relationship2D descriptorsDatasets as TopicQuantitative Structure-Activity RelationshipAntineoplastic Agents010402 general chemistry01 natural sciencesModels BiologicalAnticancer activityMLR03 medical and health sciencesInhibitory Concentration 50Drug DiscoveryLeast-Squares AnalysisTelomerase134-oxadiazolesOxadiazolesMolecular StructureDrug discoveryChemistryQSARQuantitative structureCombinatorial chemistry0104 chemical sciencesTelomerase inhibitors030104 developmental biology1 3 4 oxadiazole derivativesDrug Screening Assays AntitumorCurrent drug discovery technologies
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Stem Cell-Derived, microRNA-Carrying Extracellular Vesicles: A Novel Approach to Interfering with Mesangial Cell Collagen Production in a Hyperglycae…

2016

Extracellular vesicles (EVs) that are derived from stem cells are proving to be promising therapeutic options. We herein investigate the therapeutic potential of EVs that have been derived from different stem cell sources, bone-marrow (MSC) and human liver (HLSC), on mesangial cells (MCs) exposed to hyperglycaemia. By expressing a dominant negative STAT5 construct (ΔNSTAT5) in HG-cultured MCs, we have demonstrated that miR-21 expression is under the control of STAT5, which translates into Transforming Growth Factor beta (TGFβ) expression and collagen production. A number of approaches have been used to show that both MSC- and HLSC-derived EVs protect MCs from HG-induced damage via the trans…

0301 basic medicineMolecular biologyCellGene Expressionlcsh:MedicineBiochemistry0302 clinical medicineAnimal CellsChronic Kidney DiseaseMedicine and Health SciencesSTAT5 Transcription FactorRNA Processing Post-Transcriptionallcsh:ScienceSTAT5Energy-Producing OrganellesCells CulturedMultidisciplinarybiologyMesangial cellStem CellsVector ConstructionCell biologyMitochondriaEnzymesmedicine.anatomical_structureBiochemistryNephrology030220 oncology & carcinogenesisMesangial CellsCollagenStem cellCellular TypesCellular Structures and OrganellesOxidoreductasesLuciferaseResearch ArticleCollagen Type IVBioenergeticsDNA constructionModels Biological03 medical and health sciencesExtracellular VesiclesmicroRNAmedicineGene Expression and Vector TechniquesGeneticsHumansVesiclesCell ProliferationMolecular Biology Assays and Analysis TechniquesCell growthMesenchymal stem celllcsh:RBiology and Life SciencesProteinsMesenchymal Stem CellsTransforming growth factor betaCell BiologyResearch and analysis methodsMicroRNAs030104 developmental biologyMolecular biology techniquesGlucoseHyperglycemiabiology.proteinEnzymologylcsh:QCollagensPLoS ONE
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