Search results for "SIGNALING PATHWAY"

showing 10 items of 284 documents

WNT Signaling Perturbations Underlie the Genetic Heterogeneity of Robinow Syndrome

2018

International audience; Locus heterogeneity characterizes a variety of skeletal dysplasias often due to interacting or overlapping signaling pathways. Robinow syndrome is a skeletal disorder historically refractory to molecular diagnosis, potentially stemming from substantial genetic heterogeneity. All current known pathogenic variants reside in genes within the noncanonical Wnt signaling pathway including ROR2, WNT5A, and more recently, DVL1 and DVL3. However, ∼70% of autosomal-dominant Robinow syndrome cases remain molecularly unsolved. To investigate this missing heritability, we recruited 21 families with at least one family member clinically diagnosed with Robinow or Robinow-like pheno…

Male0301 basic medicineCandidate geneFrizzledGROWTH-PLATEDEP DOMAINlnfectious Diseases and Global Health Radboud Institute for Molecular Life Sciences [Radboudumc 4]PROTEINskeletal dysplasiaCraniofacial Abnormalities0302 clinical medicineLocus heterogeneityChromosome SegregationChild[ SDV.GEN.GH ] Life Sciences [q-bio]/Genetics/Human geneticsWnt Signaling PathwayGenetics (clinical)Genes DominantGeneticsWnt signaling pathwayMiddle AgedRobinow syndromeMENDELIAN-INHERITANCEPhenotypeChild PreschoolFemaleNEURAL-TUBE DEFECTSVERTEBRATE GASTRULATIONhuman embryonic developmentRare cancers Radboud Institute for Health Sciences [Radboudumc 9]AdultAdolescentCELL POLARITYLimb Deformities CongenitalMutation MissenseDwarfismBiologyArticledual molecular diagnosisDiagnosis DifferentialGenetic Heterogeneity03 medical and health sciencesFrizzledAll institutes and research themes of the Radboud University Medical CenterSkeletal disorderGeneticsmedicineHumansGenetic Association StudiesNeurodevelopmental disorders Donders Center for Medical Neuroscience [Radboudumc 7]Base SequenceGenetic heterogeneityMUTATIONSROR2medicine.diseaseDROSOPHILA TISSUE POLARITY030104 developmental biology[SDV.GEN.GH]Life Sciences [q-bio]/Genetics/Human geneticsUrogenital AbnormalitiesAUTOSOMAL-DOMINANT030217 neurology & neurosurgery
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Neurovascular EGFL7 regulates adult neurogenesis in the subventricular zone and thereby affects olfactory perception

2016

Adult neural stem cells reside in a specialized niche in the subventricular zone (SVZ). Throughout life they give rise to adult-born neurons in the olfactory bulb (OB), thus contributing to neural plasticity and pattern discrimination. Here, we show that the neurovascular protein EGFL7 is secreted by endothelial cells and neural stem cells (NSCs) of the SVZ to shape the vascular stem-cell niche. Loss of EGFL7 causes an accumulation of activated NSCs, which display enhanced activity and re-entry into the cell cycle. EGFL7 pushes activated NSCs towards quiescence and neuronal progeny towards differentiation. This is achieved by promoting Dll4-induced Notch signalling at the blood vessel-stem …

Male0301 basic medicineGeneral Physics and AstronomyNEURAL STEM-CELLSMOUSEMiceSUBEPENDYMAL ZONENeural Stem CellsLateral VentriclesLINEAGE PROGRESSIONBRAININ-VIVOMice KnockoutNeuronal PlasticityMultidisciplinaryCell CycleQNeurogenesisNICHEAnatomyNeural stem cellCell biologyAdult Stem Cellsmedicine.anatomical_structureSignal TransductionSTIMULATES NEUROGENESISEGF Family of ProteinsNeurogenesisScienceNotch signaling pathwaySubventricular zoneBiologyInhibitory postsynaptic potentialArticleGeneral Biochemistry Genetics and Molecular Biology03 medical and health sciencesNeuroplasticitymedicineBiological neural networkAnimalsCalcium-Binding ProteinsProteinsGeneral ChemistryOlfactory PerceptionENDOTHELIAL-CELLSnervous system diseasesOlfactory bulbMice Inbred C57BLSELF-RENEWAL030104 developmental biologynervous system
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Molecular mechanisms mediating the neuroprotective role of the selective estrogen receptor modulator, bazedoxifene, in acute ischemic stroke: A compa…

2017

As the knowledge on the estrogenic system in the brain grows, the possibilities to modulate it in order to afford further neuroprotection in brain damaging disorders so do it. We have previously demonstrated the ability of the selective estrogen receptor modulator, bazedoxifene (BZA), to reduce experimental ischemic brain damage. The present study has been designed to gain insight into the molecular mechanisms involved in such a neuroprotective action by investigating: 1) stroke-induced apoptotic cell death; 2) expression of estrogen receptors (ER) ERα, ERβ and the G-protein coupled estrogen receptor (GPER); and 3) modulation of MAPK/ ERK1/2 and PI3K/Akt signaling pathways. For comparison, …

Male0301 basic medicineMAPK/ERK pathwayIndolesSignaling pathwaysEndocrinology Diabetes and MetabolismClinical BiochemistryEstrogen receptorApoptosisEstrogen receptorsSecond Messenger SystemsBiochemistryBrain IschemiaReceptors G-Protein-Coupled0302 clinical medicineEndocrinologyPhosphatidylinositol PhosphatesCerebral CortexNeuronsEstradiolNeuroprotectionStrokeNeuroprotective AgentsSelective estrogen receptor modulatorReperfusion InjuryMolecular MedicineSelective estrogen receptor modulatorsGPERmedicine.medical_specialtyMAP Kinase Signaling Systemmedicine.drug_classAcute ischemic strokeNerve Tissue ProteinsBazedoxifeneBiologyNeuroprotection03 medical and health sciencesInternal medicinemedicineAnimalsEstrogen Receptor betaRats WistarMolecular BiologyProtein kinase BPI3K/AKT/mTOR pathwayEstrogen Receptor alphaEstrogensCell BiologyEstrogen030104 developmental biologyEndocrinologyEstrogen030217 neurology & neurosurgeryThe Journal of Steroid Biochemistry and Molecular Biology
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The canonical but not the noncanonical wnt pathway inhibits the development of allergic airway disease

2018

Abstract Asthma is a syndrome with multifactorial causes, resulting in a variety of different phenotypes. Current treatment options are not curative and are sometimes ineffective in certain disease phenotypes. Therefore, novel therapeutic approaches are required. Recent findings have shown that activation of the canonical Wnt signaling pathway suppresses the development of allergic airway disease. In contrast, the effect of the noncanonical Wnt signaling pathway activation on allergic airway disease is not well described. The aim of this study was to validate the therapeutic effectiveness of Wnt-1–driven canonical Wnt signaling compared with Wnt-5a–driven noncanonical signaling in murine mo…

Male0301 basic medicineOvalbuminT-LymphocytesT cellImmunologyMedizinWnt1 ProteinLymphocyte ActivationWnt-5a ProteinImmunomodulationMice03 medical and health sciences0302 clinical medicineImmune systemMetaplasiaRespiratory HypersensitivitymedicineAnimalsHumansImmunology and AllergyWnt Signaling PathwayCells CulturedMice Inbred BALB CGoblet cellbusiness.industryWnt signaling pathwayDendritic CellsAllergensrespiratory systemPhenotypeAsthmaIn vitroMice Inbred C57BLDisease Models Animal030104 developmental biologymedicine.anatomical_structureCancer researchFemalemedicine.symptombusinessFunction (biology)030215 immunology
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Activated Thyroid Hormone Promotes Differentiation and Chemotherapeutic Sensitization of Colorectal Cancer Stem Cells by Regulating Wnt and BMP4 Sign…

2016

Abstract Thyroid hormone is a pleiotropic factor that controls many cellular processes in multiple cell types such as cancer stem cells (CSC). Thyroid hormone concentrations in the blood are stable, but the action of the deiodinases (D2–D3) provides cell-specific regulation of thyroid hormone activity. Deregulation of deiodinase function and thyroid hormone status has been implicated in tumorigenesis. Therefore, we investigated the role of thyroid hormone metabolism and signaling in colorectal CSCs (CR-CSC), where deiodinases control cell division and chemosensitivity. We found that increased intracellular thyroid hormone concentration through D3 depletion induced cell differentiation and s…

Male0301 basic medicineThyroid Hormonesendocrine systemCancer Researchmedicine.medical_specialtyendocrine system diseasesCellular differentiationDeiodinaseBone Morphogenetic Protein 4Colorectal NeoplasmMice03 medical and health sciencesCancer stem cellCell Line TumorInternal medicinemedicineAnimalsHumansThyroid HormoneWnt Signaling PathwayHormone activityThyroid hormone receptorbiologyAnimalThyroidWnt signaling pathwayCell DifferentiationMiddle Aged030104 developmental biologyEndocrinologymedicine.anatomical_structureOncologyNeoplastic Stem CellsCancer researchbiology.proteinNeoplastic Stem CellColorectal NeoplasmsHumanSignal TransductionHormoneCancer Research
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Astrocytic Calcium Waves Signal Brain Injury to Neural Stem and Progenitor Cells

2017

Summary Brain injuries, such as stroke or trauma, induce neural stem cells in the subventricular zone (SVZ) to a neurogenic response. Very little is known about the molecular cues that signal tissue damage, even over large distances, to the SVZ. Based on our analysis of gene expression patterns in the SVZ, 48 hr after an ischemic lesion caused by middle cerebral artery occlusion, we hypothesized that the presence of an injury might be transmitted by an astrocytic traveling calcium wave rather than by diffusible factors or hypoxia. Using a newly established in vitro system we show that calcium waves induced in an astrocytic monolayer spread to neural stem and progenitor cells and increase th…

Male0301 basic medicineTime FactorsNotch signaling pathwaySubventricular zonechemistry.chemical_elementBiologyCalciumcalcium signalingBiochemistryArticleMice03 medical and health sciencesAdenosine TriphosphateNeural Stem CellsDownregulation and upregulationCell MovementGeneticsmedicineAnimalsCell Self RenewalProgenitor celllcsh:QH301-705.5Cells CulturedCalcium signalinglcsh:R5-920Gene Expression ProfilingastrocytesGap JunctionsCell DifferentiationCell BiologyAnatomyHypoxia (medical)strokeNeural stem cellDisease Models Animal030104 developmental biologymedicine.anatomical_structurelcsh:Biology (General)chemistryBrain InjuriesCalciummedicine.symptomFunction and Dysfunction of the Nervous Systemlcsh:Medicine (General)TranscriptomeNeurosciencenotchDevelopmental BiologyStem Cell Reports
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WNT2b activates epithelial-mesenchymal transition through FZD4: relevance in penetrating Crohns disease.

2020

Abstract Background and Aims Epithelial-mesenchymal transition [EMT] has been related to fibrosis and fistula formation, common complications associated with Crohn´s disease [CD]. The WNT signalling pathway mediates EMT, and specific WNT/FZD interactions have been related to the activation of this process in several diseases. We aim to analyse the relevance of EMT and WNT ligands and receptors in the penetrating behaviour of CD. Methods Intestinal surgical resections were obtained from control and CD patients with a stenotic or penetrating behaviour. Fibrosis was determined by the histological analysis of collagen deposition and EMT by confocal microscopy. The expression of WNT ligands, inh…

Male0301 basic medicineWNT pathwayVimentin0302 clinical medicineCrohn DiseaseFibrosisMedicineIntestinal MucosaReceptorWnt Signaling PathwayAged 80 and overbiologyGastroenterologyWnt signaling pathwayGeneral MedicineMiddle AgedCrohn's disease10219 Clinic for Gastroenterology and Hepatology030220 oncology & carcinogenesisembryonic structuresFemaleHT29 CellsAdultEpithelial-Mesenchymal TransitionAdolescentColonBlotting Western610 Medicine & healthReal-Time Polymerase Chain ReactionYoung Adult03 medical and health sciencesHT29 CellsHumansImmunoprecipitation2715 GastroenterologyEpithelial–mesenchymal transitionCrohn´s disease WNT pathway fibrosisAgedGlycoproteinsCadherinbusiness.industryfibrosismedicine.diseaseFibrosisFrizzled ReceptorsIn vitroWnt Proteins030104 developmental biologyCancer researchbiology.proteinbusiness
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Can the TLR-4-mediated signaling pathway be "a key inflammatory promoter for sporadic TAA"?

2014

Thoracic aorta shows with advancing age various changes and a progressive deterioration in structure and function. As a result, vascular remodeling (VR) and medial degeneration (MD) occur as pathological entities responsible principally for the sporadic TAA onset. Little is known about their genetic, molecular, and cellular mechanisms. Recent evidence is proposing the strong role of a chronic immune/inflammatory process in their evocation and progression. Thus, we evaluated the potential role of Toll like receptor- (TLR-) 4-mediated signaling pathway and its polymorphisms in sporadic TAA. Genetic, immunohistochemical, and biochemical analyses were assessed. Interestingly, the rs4986790 TLR4…

MaleArticle SubjectGenotypeImmunologyAortic DiseasesSettore MED/41 - AnestesiologiaSingle-nucleotide polymorphismAorta ThoracicSettore MED/08 - Anatomia PatologicaBiologyPolymorphism Single NucleotideImmune systemPolymorphism (computer science)Genotypelcsh:PathologySettore MED/05 - Patologia ClinicaHumansGenetic Predisposition to DiseaseAgedToll-like receptorPolymorphism GeneticSettore MED/23 - Chirurgia CardiacaCell BiologyMiddle AgedPhenotypeImmunohistochemistryToll-Like Receptor 4medial degeneration sporadic thoracic aortic aneurysm TLR-4 mediated signaling pathway rs4986790 TLR4 polymorphism translation of genetic immunohistochemical and biochemical data clinical practiceImmunologyTLR4Matrix Metalloproteinase 2FemaleSignal transductionlcsh:RB1-214Research ArticleSignal Transduction
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Clobetasol promotes neuromuscular plasticity in mice after motoneuronal loss via sonic hedgehog signaling, immunomodulation and metabolic rebalancing

2021

AbstractMotoneuronal loss is the main feature of amyotrophic lateral sclerosis, although pathogenesis is extremely complex involving both neural and muscle cells. In order to translationally engage the sonic hedgehog pathway, which is a promising target for neural regeneration, recent studies have reported on the neuroprotective effects of clobetasol, an FDA-approved glucocorticoid, able to activate this pathway via smoothened. Herein we sought to examine functional, cellular, and metabolic effects of clobetasol in a neurotoxic mouse model of spinal motoneuronal loss. We found that clobetasol reduces muscle denervation and motor impairments in part by restoring sonic hedgehog signaling and …

MaleCancer ResearchPhysiology129 StrainBiochemistryMiceDatabases GeneticMedicineMyocyteMotor NeuronsNeuronal PlasticitySkeletalSmoothened ReceptorHedgehog signaling pathwayMuscle atrophyMitochondriaAstrogliosisNeuroprotective AgentsMusclemedicine.symptomInflammation MediatorsSignal TransductionCholera ToxinMice 129 StrainhedgehogImmunologyMotor ActivityNeuroprotectionArticleDatabasesCellular and Molecular NeurosciencesmoothenedGeneticAnimalsHumansHedgehog ProteinsMuscle SkeletalHedgehogGlucocorticoidsMuscle DenervationQH573-671Animalbusiness.industryAmyotrophic Lateral SclerosisGlial biologyCell Biologymedicine.diseaseSaporinsSpineMitochondria MuscleDisease Models AnimalclobetasolinflammationCase-Control StudiesDisease ModelsDiseases of the nervous systemCytologySmoothenedbusinessEnergy MetabolismNeuroscienceOpen Field Test
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Loss of function mutation in the palmitoyl-transferase HHAT leads to syndromic 46,XY disorder of sex development by impeding Hedgehog protein palmito…

2014

The Hedgehog (Hh) family of secreted proteins act as morphogens to control embryonic patterning and development in a variety of organ systems. Post-translational covalent attachment of cholesterol and palmitate to Hh proteins are critical for multimerization and long range signaling potency. However, the biological impact of lipid modifications on Hh ligand distribution and signal reception in humans remains unclear. In the present study, we report a unique case of autosomal recessive syndromic 46,XY Disorder of Sex Development (DSD) with testicular dysgenesis and chondrodysplasia resulting from a homozygous G287V missense mutation in the hedgehog acyl-transferase (HHAT) gene. This mutation…

MaleCancer Research[SDV]Life Sciences [q-bio]medicine.disease_causeCell Fate DeterminationMiceTestisMorphogenesisMissense mutationddc:576.5Genetics (clinical)MutationHomozygoteCell DifferentiationHedgehog signaling pathwayPedigreeCell biologyFemaleSignal transductionSignal TransductionResearch Articlemedicine.medical_specialtylcsh:QH426-470LipoylationMolecular Sequence DataMutation MissenseBiologyPalmitoylationHHATInternal medicineGeneticsmedicineAnimalsHumansHedgehog ProteinsAmino Acid SequenceMolecular BiologyHedgehogEcology Evolution Behavior and SystematicsDisorder of Sex Development 46XY[ SDV ] Life Sciences [q-bio]Sequence Homology Amino AcidBiology and Life SciencesSex Determinationlcsh:GeneticsEndocrinology46 XY Disorders of Sex Development/*genetics; Acyltransferases/chemistry/*genetics/metabolism; Amino Acid Sequence; Animals; Female; Hedgehog Proteins/*metabolism; Homozygote; Humans; Lipoylation/*genetics; Male; Mice; Molecular Sequence Data; *Mutation Missense; Pedigree; Sequence Homology Amino Acid; Signal Transduction/*genetics; Testis/embryologyLipid modificationAcyltransferasesDevelopmental Biology
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