Search results for "Recess"

showing 10 items of 379 documents

Maternal Inheritance of a Recessive RBP4 Defect in Canine Congenital Eye Disease

2018

SUMMARY Maternally skewed transmission of traits has been associated with genomic imprinting and oocyte-derived mRNA. We report canine congenital eye malformations, caused by an amino acid deletion (K12del) near the N terminus of retinol-binding protein (RBP4). The disease is only expressed when both dam and offspring are deletion homozygotes. RBP carries vitamin A (retinol) from hepatic stores to peripheral tissues, including the placenta and developing eye, where it is required to synthesize retinoic acid. Gestational vitamin A deficiency is a known risk factor for ocular birth defects. The K12del mutation disrupts RBP folding in vivo, decreasing its secretion from hepatocytes to serum. T…

0301 basic medicineMaleNon-Mendelian inheritanceProtein Foldingcongenital eye defectEye Diseasesgenetic structuresNATIVE DISULFIDE BONDSMedical PhysiologyRetinoic acidReproductive health and childbirth413 Veterinary scienceMicrophthalmiavitamin Achemistry.chemical_compoundPlasmaA-vitamiini2.1 Biological and endogenous factorsMicrophthalmosPrealbuminCRYSTAL-STRUCTUREAetiologyBase Pairinglcsh:QH301-705.5Sequence DeletionPediatricwhole genome sequencingVITAMIN-A-DEFICIENCYANOPHTHALMIAPenetrancePedigreemedicine.anatomical_structurePhenotypeFemalemedicine.medical_specialtyGenotypeENDOPLASMIC-RETICULUMGenes RecessiveMETABOLISMBiologyGeneral Biochemistry Genetics and Molecular BiologyArticle03 medical and health sciencesDogscanine geneticsInternal medicinePlacentaRETINOL-BINDING-PROTEINGeneticsmedicineAnimalsHumansRecessiveMALFORMATIONSBIOCHEMICAL BASISAmino Acid SequenceAlleleEye Disease and Disorders of VisionNutritiongenome-wide association study030102 biochemistry & molecular biologywestern blottingMUTATIONSta1184RBP4maternal inheritancemedicine.diseaseRetinol-Binding ProteinsRetinol binding proteinnuclear magnetic resonance030104 developmental biologyEndocrinologychemistryGeneslcsh:Biology (General)microphthalmiaGenetic LociHela Cells1182 Biochemistry cell and molecular biologyCongenital Structural Anomalies3111 BiomedicineBiochemistry and Cell BiologyDigestive DiseasesGenomic imprintingRetinol-Binding Proteins PlasmaHeLa Cells
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Genetic epidemiology of autosomal recessive hypercholesterolemia in Sicily: Identification by next-generation sequencing of a new kindred.

2017

Background Autosomal recessive hypercholesterolemia (ARH) is a rare inherited lipid disorder. In Sardinia, differently from other world regions, the mutated allele frequency is high. It is caused by mutations in the low-density lipoprotein receptor adaptor protein 1 gene. Fourteen different mutations have been reported so far; in Sardinia, 2 alleles (ARH1 and ARH2) explain most of the cases. Four ARH patients, all carriers of the ARH1 mutation, have been identified in mainland Italy and 2 in Sicily. Objective The objectives of the study were to improve the molecular diagnosis of familial hypercholesterolemia (FH) and to estimate the frequency of the ARH1 allele in 2 free-living Sicilian pop…

0301 basic medicineMaleSettore MED/09 - Medicina InternaEndocrinology Diabetes and MetabolismFamilial hypercholesterolemia030204 cardiovascular system & hematology0302 clinical medicineChildN-Glycosyl HydrolasesSicilyGeneticsAged 80 and overeducation.field_of_studyNutrition and DieteticsAllele frequencyHomozygoteHigh-Throughput Nucleotide SequencingAutosomal recessive hypercholesterolemiaMiddle AgedAutosomal Recessive HypercholesterolemiaSettore MED/26 - NeurologiaFemaleCardiology and Cardiovascular MedicineAdultAdolescentGenotypePopulationHypercholesterolemiaBiologyDNA sequencing03 medical and health sciencesYoung AdultARH1Internal MedicinemedicineHumansAlleleeducationGenotypingAllele frequencyAllelesAdaptor Proteins Signal TransducingAgedHeterozygous carrierSequence Analysis DNAmedicine.diseaseNGS-based gene panel030104 developmental biologyGenetic epidemiologyReceptors LDLJournal of clinical lipidology
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PDXK mutations cause polyneuropathy responsive to pyridoxal 5′‐phosphate supplementation

2019

OBJECTIVE: To identify disease-causing variants in autosomal recessive axonal polyneuropathy with optic atrophy and provide targeted replacement therapy. METHODS: We performed genome-wide sequencing, homozygosity mapping, and segregation analysis for novel disease-causing gene discovery. We used circular dichroism to show secondary structure changes and isothermal titration calorimetry to investigate the impact of variants on adenosine triphosphate (ATP) binding. Pathogenicity was further supported by enzymatic assays and mass spectroscopy on recombinant protein, patient-derived fibroblasts, plasma, and erythrocytes. Response to supplementation was measured with clinical validated rating sc…

0301 basic medicineMale[SDV.NEU.NB]Life Sciences [q-bio]/Neurons and Cognition [q-bio.NC]/NeurobiologyLOCAL TRANSLATIONMedizinmedicine.disease_causeDISEASEchemistry.chemical_compound0302 clinical medicinepolineuropathyCinètica enzimàticaGene Regulatory NetworksPyridoxal phosphateChildPyridoxal KinaseAdenosine triphosphate (ATP)Research ArticlesAged 80 and overMutationGene Regulatory NetworkPLASMAAutosomal recessive axonal polyneuropathyDisease gene identificationPyridoxal kinase3. Good healthSettore MED/26 - NEUROLOGIANeuropaties perifèriquesTreatment OutcomePolyneuropathieNeurologyChild PreschoolPyridoxal PhosphateRELIABILITYVitamin B ComplexFemaleLife Sciences & BiomedicinePolyneuropathyHumanResearch ArticleAdultAdolescentPDXKClinical NeurologyCHARCOT-MARIE-TOOTHCHARCOT-MARIE-TOOTH CMT NEUROPATHY SCORE LOCAL TRANSLATION DISEASE RELIABILITY; MECHANISMS DISCOVERY FRAMEWORK KINASE PLASMAMECHANISMS03 medical and health sciencesPolyneuropathiesAtrophy[SDV.BBM.GTP]Life Sciences [q-bio]/Biochemistry Molecular Biology/Genomics [q-bio.GN]KINASEmedicineHumansCMT NEUROPATHY SCOREPDXK mutationsPyridoxalDietary SupplementAgedPeripheral neuropathiesScience & Technology[SCCO.NEUR]Cognitive science/NeuroscienceEnzyme kineticsNeurosciencesFRAMEWORKmedicine.diseaseMolecular biology030104 developmental biologychemistryDISCOVERYDietary SupplementsMutationNeurosciences & NeurologyNeurology (clinical)Adenosine triphosphate030217 neurology & neurosurgeryAnnals of Neurology
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Extending the ALDH18A1 clinical spectrum to severe autosomal recessive fetal cutis laxa with corpus callosum agenesis

2018

IF 3.822 (2018); International audience

0301 basic medicinePathologymedicine.medical_specialtyFetusALDH18A1Corpus Callosum Agenesisbusiness.industryGenes RecessiveAldehyde Dehydrogenase030105 genetics & hereditymedicine.diseaseMagnetic Resonance ImagingCutis Laxa03 medical and health sciencesFetus[SDV.GEN.GH]Life Sciences [q-bio]/Genetics/Human geneticsGeneticsmedicineHumansAgenesis of Corpus CallosumbusinessAllelesGenetics (clinical)Cutis laxa
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Recession Coverage Using Soft Tissue Substitutes

2018

Today, a variety of surgical procedures can be used to successfully treat gingival recession defects. Among them, the autogenous connective tissue graft in conjunction with a coronally advanced flap is commonly considered the gold standard procedure. However, the most significant disadvantages of this procedure are the potential morbidity associated with autogenous tissue harvesting and the limited availability of donor tissue. For these reasons, alternative surgical procedures using membranes, enamel matrix derivative, and soft tissue graft substitutes have been proposed and tested. The aim of the present chapter is to provide an overview on the use of soft tissue substitutes as a possible…

0301 basic medicinebusiness.industryDonor tissueDentistryConnective tissueSoft tissue030206 dentistrySurgical proceduresSoft tissue graft03 medical and health sciences030104 developmental biology0302 clinical medicinemedicine.anatomical_structureTissue HarvestingEnamel matrix derivativeMedicinemedicine.symptombusinessGingival recession
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Coronally advanced flap and connective tissue graft with or without plasma rich in growth factors (PRGF) in treatment of gingival recession

2017

Background Several researchers have tried to improve the results of gingival recession treatment techniques. One of the methods is to use growth factors The present study was undertaken to evaluate the effect of CAF (coronally advanced flap) + CTG (connective tissue graft) + PRGF (plasma rich in growth factors) in the treatment of Miller Class I buccal gingival recession. Material and methods Twenty-two teeth with Miller Class I gingival recession in 6 patients 26 ‒ 47 years of age were included in a split-mouth designed randomized controlled trial (RCT). In each patient, one side was treated with CAF + CTG + PRGF (test) and the other side was treated with CAF + CTG (control). The following…

0301 basic medicinemedicine.medical_specialtyMucogingival junctionbusiness.industryResearchConnective tissueAttachment levelRepeated measures designDentistryMucogingival surgery030206 dentistry:CIENCIAS MÉDICAS [UNESCO]Root coverage03 medical and health sciencesPlastic surgery030104 developmental biology0302 clinical medicinemedicine.anatomical_structureUNESCO::CIENCIAS MÉDICASmedicinePeriodontologymedicine.symptombusinessGeneral DentistryGingival recession
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Autosomal Recessive Hypercholesterolemia

2018

Abstract Background Autosomal recessive hypercholesterolemia (ARH) is a rare lipid disorder characterized by premature atherosclerotic cardiovascular disease (ASCVD). There are sparse data for clinical management and cardiovascular outcomes in ARH. Objectives Evaluation of changes in lipid management, achievement of low-density lipoprotein cholesterol (LDL-C) goals and cardiovascular outcomes in ARH. Methods Published ARH cases were identified by electronic search. All corresponding authors and physicians known to treat these patients were asked to provide follow-up information, using a standardized protocol. Results We collected data for 52 patients (28 females, 24 males; 31.1 ± 17.1 years…

0301 basic medicinemedicine.medical_specialtyStatinAtherosclerotic cardiovascular diseasebusiness.industrymedicine.drug_class030204 cardiovascular system & hematologyLomitapide03 medical and health scienceschemistry.chemical_compound030104 developmental biology0302 clinical medicineEzetimibechemistryAutosomal Recessive HypercholesterolemiaInternal medicinemedicineEffective treatmentlipids (amino acids peptides and proteins)In patientCardiology and Cardiovascular MedicinebusinessCardiovascular outcomesmedicine.drugJournal of the American College of Cardiology
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The language of recovery

2020

Abstract The present study attempts to make a comparative analysis of two Spanish and American political speeches, which belong to two different debate traditions, in terms of the metaphors used. For that purpose, we analyze the Economy sections of the 2015 State of the Union Address in the US and in the 2015 State of the Nation Debate in Spain. The present study aims at answering the following research questions: What metaphors do President Obama and Prime Minister Rajoy use in the American and Spanish political speeches to convince their audiences of America’s and Spain’s economic victory? What are the similarities and differences between the representations depicted by metaphor use in th…

050101 languages & linguisticsLinguistics and LanguageMetaphormedia_common.quotation_subject05 social sciencesVictoryMedia studiesReification (fallacy)Recession050601 international relationsLanguage and Linguistics0506 political sciencePoliticsState (polity)Political scienceEconomic recovery0501 psychology and cognitive sciencesNew economymedia_commonRevista Española de Lingüística Aplicada/Spanish Journal of Applied Linguistics
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Revolving doors: are they valued in the Spanish stock market?

2016

Purpose The purpose of this paper is to examine the effect of political connections in the value of companies listed on the Spanish stock market. Design/methodology/approach The authors study two issues on this topic: the impact on the company value of the appointment as director of an ex-politician through an event study; and the long run stock market performance of companies with political connections. The authors employ a sample of listed firms in the Spanish stock market for which the authors collected data about their political connections. The authors perform the study during a wide period (1996-2011), comprising four legislatures. Findings The results show that from the market persp…

050208 financePublic AdministrationTransparency (market)business.industryStrategy and Managementmedia_common.quotation_subject05 social sciencesEvent studyAccountingLegislatureRecessionPoliticsGood governance0502 economics and businessValue (economics)EconomicsStock market050207 economicsBusiness and International ManagementMarketingbusinessGeneral Economics Econometrics and Financemedia_commonAcademia Revista Latinoamericana de Administración
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Accretion in strong field gravity with eXTP

2019

In this paper we describe the potential of the enhanced X-ray Timing and Polarimetry (eXTP) mission for studies related to accretion flows in the strong field gravity regime around both stellar-mass and supermassive black-holes. eXTP has the unique capability of using advanced 'spectral-timing-polarimetry' techniques to analyze the rapid variations with three orthogonal diagnostics of the flow and its geometry, yielding unprecedented insight into the inner accreting regions, the effects of strong field gravity on the material within them and the powerful outflows which are driven by the accretion process.

ACTIVE GALACTIC NUCLEIAccretionaccretion; black holes physics; X-ray; Physics and Astronomy (all)black holes physicAstronomyAstrophysics::High Energy Astrophysical PhenomenaBlack holes physicsPolarimetryFOS: Physical sciencesBLACK-HOLE SPINGeneral Physics and AstronomyStrong fieldAstrophysicsAstrophysics::Cosmology and Extragalactic Astrophysics01 natural sciencesX-rayPhysics and Astronomy (all)ELECTROMAGNETIC EMISSIONSettore FIS/05 - Astronomia e Astrofisicablack holes physicsaccretion0103 physical sciencesAstrophysics::Solar and Stellar Astrophysics010306 general physics010303 astronomy & astrophysicsAstrophysics::Galaxy AstrophysicsHigh Energy Astrophysical Phenomena (astro-ph.HE)XMM-NEWTONPhysicsLENS-THIRRING PRECESSION[SDU.ASTR]Sciences of the Universe [physics]/Astrophysics [astro-ph]QUASI-PERIODIC OSCILLATIONS[SDU.ASTR.HE]Sciences of the Universe [physics]/Astrophysics [astro-ph]/High Energy Astrophysical Phenomena [astro-ph.HE]IRON KAccretion (astrophysics)X ray[SDU]Sciences of the Universe [physics]ULTRA-FAST OUTFLOWSAstrophysics::Earth and Planetary AstrophysicsSPECTRAL FEATURESAstrophysics - High Energy Astrophysical Phenomena[PHYS.ASTR]Physics [physics]/Astrophysics [astro-ph]X-RAY BINARIESScience China Physics, Mechanics & Astronomy
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