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showing 10 items of 819 documents

TCTN3 Mutations Cause Mohr-Majewski Syndrome

2012

Orofaciodigital syndromes (OFDSs) consist of a group of heterogeneous disorders characterized by abnormalities in the oral cavity, face, and digits and associated phenotypic abnormalities that lead to the delineation of 13 OFDS subtypes. Here, by a combined approach of homozygozity mapping and exome ciliary sequencing, we identified truncating TCTN3 mutations as the cause of an extreme form of OFD associated with bone dysplasia, tibial defect, cystic kidneys, and brain anomalies (OFD IV, Mohr-Majewski syndrome). Analysis of 184 individuals with various ciliopathies (OFD, Meckel, Joubert, and short rib polydactyly syndromes) led us to identify four additional truncating TCTN3 mutations in un…

AdolescentFoot Deformities CongenitalMolecular Sequence DataCiliopathiesJoubert syndromeYoung AdultFetusReportCerebellumGLI3medicineGeneticsHumansExomeHedgehog ProteinsGenetics(clinical)Sonic hedgehogChildExomeGenetics (clinical)Adaptor Proteins Signal TransducingCystic kidneyGeneticsBase SequencebiologyHomozygoteIntracellular Signaling Peptides and ProteinsMembrane ProteinsCiliary transition zoneSequence Analysis DNAOrofaciodigital Syndromesmedicine.diseaseCleft PalateCiliopathyPhenotypeMutationbiology.proteinApoptosis Regulatory ProteinsHand Deformities CongenitalSignal TransductionThe American Journal of Human Genetics
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A recurrent TP63 mutation causing EEC3 and Rapp–Hodgkin syndromes

2016

The ectrodactyly, ectodermal dysplasia, and cleft lip/palate syndrome 3 (EEC3; OMIM #604292), the Rapp-Hodgkin syndrome (RHS), and various other syndromes are caused by mutations in the TP63 gene, which encodes a p53-like transcription factor. Here, we report on a woman aged 37 years and her daughter aged 3 years with the previously reported c.1028G>A (p.Arg343Gln) mutation in exon 8 of TP63. The mother lacked ectrodactyly, indicating a diagnosis of RHS, whereas the girl presented with all three major features (ectrodactyly, ectodermal dysplasia, clefting) and different minor features (including small and brittle nails, and recurrent conjunctivitis believed to be because of stenotic and blo…

Adult0301 basic medicineHeterozygoteEctodermal dysplasiamedicine.medical_specialtyEctrodactylyFoot Deformities CongenitalCleft Lipmedia_common.quotation_subjectmedicine.disease_causePathology and Forensic MedicineFingers030207 dermatology & venereal diseases03 medical and health sciencesExon0302 clinical medicineEctodermal DysplasiaTP63medicineHumansAlleleAllelesGenetics (clinical)media_commonDaughterMutationbusiness.industryTumor Suppressor ProteinsFaciesExonsGeneral Medicinemedicine.diseaseDermatologyPenetrancePedigreeCleft PalatePhenotype030104 developmental biologyAmino Acid SubstitutionChild PreschoolMutationPediatrics Perinatology and Child HealthFemaleAnatomybusinessHand Deformities CongenitalTranscription FactorsClinical Dysmorphology
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Normal values of M mode echocardiographic measurements of more than 2000 healthy infants and children in central Europe

2000

OBJECTIVE—To obtain normal M mode (one dimensional) echocardiographic values in a substantial sample of normal infants and children.
DESIGN—Data were obtained over three years from a single centre in central Europe.
PATIENTS—2036 healthy infants and children aged one day to 18 years.
METHODS—In line with recommendations for standardising measurements from M mode echocardiograms, and using digital echocardiographic equipment, measurements were obtained of the following: right ventricular anterior wall thickness at end diastole, right ventricular end diastolic dimension, thickness of interventricular septum at end diastole and end systole, thickness of posterior wall of the left ventricle at …

AdultAortic valvemedicine.medical_specialtyAdolescentBody Surface AreaAnterior wallDiastoleNormal valuesCardiovascular MedicineReference ValuesLeft atrialInternal medicineHumansMedicineInterventricular septumChildBody surface areabusiness.industryInfant NewbornInfantmedicine.anatomical_structureEchocardiographyVentricleChild PreschoolCardiologyRegression AnalysisCardiology and Cardiovascular MedicinebusinessHeart
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The impact of orthognathic surgery on quality of life in individuals with oral clefts

2021

Summary Background/objectives To evaluate the relationships between individual, environmental, clinical factors and oral health-related quality of life (OHRQoL) in patients with cleft lip and palate (CLP) following orthognathic surgery. Materials and methods A follow-up study was conducted involving 69 adults with unilateral and bilateral CLP under orthodontic treatment. Interviews and oral examinations were conducted prior to orthognathic surgery (T0) to evaluate age, gender, psychological well-being, dental caries, malocclusion, social support, social networks, family income and education and OHRQoL. All participants were reviewed after 6 months (T1) to re-assess psychological well-being,…

AdultBiopsychosocial modelCleft Lipmedicine.medical_treatmentOrthognathic surgeryDentistryOral HealthOrthodonticsDental Caries03 medical and health sciencesSocial support0302 clinical medicineQuality of lifeSurveys and QuestionnairesFISSURA LÁBIOPALATINAmedicineHumansIn patientProspective Studies030223 otorhinolaryngologyProspective cohort studybusiness.industryOrthognathic Surgery030206 dentistrymedicine.diseasehumanitiesCleft PalateQuality of LifeOral examinationMalocclusionbusinessMalocclusionFollow-Up StudiesEuropean Journal of Orthodontics
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Hemostatic function in young subjects with central obesity: relationship with left ventricular function.

1995

This study was designed to evaluate coagulation and fibrinolysis activity and their relationship with left ventricular function in young obese subjects with central fat distribution. We assessed coagulation and fibrinolysis activity by evaluation of factor VII activity, fibrinogen and plasminogen, plasminogen activator inhibitor (PAI), and tissue plasminogen activator antigen basally (tPA1) and after venous occlusion (tPA2). These measures were evaluated in young (< 40 years) obese subjects with central fat distribution (n = 19) and in comparable lean subjects (n = 20). Blood glucose, triglycerides, total and high-density lipoprotein (HDL) cholesterol, apolipoprotein (apo) A1 and apo B, fas…

AdultBlood GlucoseMalemedicine.medical_specialtySettore MED/09 - Medicina InternaApolipoprotein BEndocrinology Diabetes and Metabolismmedicine.medical_treatmentFibrinogenVentricular Function LeftSettore MED/15 - Malattie Del Sanguechemistry.chemical_compoundEndocrinologyWaist–hip ratioInternal medicineFibrinolysismedicineHumansInsulinObesityHemostatic functionBlood CoagulationApolipoproteins ATriglyceridesApolipoproteins BHemostasisbiologybusiness.industryCholesterolCholesterol HDLFibrinogenCentral obesity Hemostatic function left ventricular functionPlasminogenFactor VIISettore MED/11 - Malattie Dell'Apparato CardiovascolarePlasminogen InactivatorsEndocrinologychemistrybiology.proteinBody ConstitutionRegression AnalysisFemalebusinessPlasminogen activatormedicine.drugLipoproteinMetabolism: clinical and experimental
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MIC trial: metoprolol in patients with mild to moderate heart failure: effects on ventricular function and cardiopulmonary exercise testing

2000

Beta-blocker therapy results in a functional benefit in patients with heart failure (CHF) due to idiopathic dilated cardiomyopathy (DCM). This study assessed if similar effects were observed in patients with ischemic heart disease (CAD), NYHA II–III after 6 months of therapy with metoprolol. Methods and results: Fifty-two patients with CHF secondary to DCM (26 patients) and CAD (26 patients) and a left ventricular ejection fraction (EF) < 40% were enrolled in the placebo-controlled study. The study medication was titrated over 6 weeks, the mean final dosage was 135 mg/day. Three patients died due to cardiogenic shock, two received placebo and one metoprolol. Eight patients did not complete …

AdultCardiomyopathy DilatedMaleCardiac function curvemedicine.medical_specialtyCardiac VolumeAdrenergic beta-AntagonistsMyocardial IschemiaCardiomyopathyVentricular Function LeftDouble-Blind MethodInternal medicineIdiopathic dilated cardiomyopathyHeart ratemedicineHumansProspective Studiescardiovascular diseasesAgedMetoprololHeart FailureEjection fractionbusiness.industryCardiogenic shockMiddle Agedmedicine.diseaseHeart failureExercise Testcardiovascular systemCardiologyFemaleCardiology and Cardiovascular Medicinebusinesshuman activitiesMetoprololcirculatory and respiratory physiologymedicine.drugEuropean Journal of Heart Failure
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Association of Left Ventricular Systolic Dysfunction Among Carriers of Truncating Variants in Filamin C With Frequent Ventricular Arrhythmia and End-…

2021

Importance: Truncating variants in the gene encoding filamin C (FLNCtv) are associated with arrhythmogenic and dilated cardiomyopathies with a reportedly high risk of ventricular arrhythmia.Objective: To determine the frequency of and risk factors associated with adverse events among FLNCtv carriers compared with individuals carrying TTN truncating variants (TTNtv).Design, Setting, and Participants: This cohort study recruited 167 consecutive FLNCtv carriers and a control cohort of 244 patients with TTNtv matched for left ventricular ejection fraction (LVEF) from 19 European cardiomyopathy referral units between 1990 and 2018. Data analyses were conducted between June and October, 2020.Main…

AdultCardiomyopathy DilatedMalemedicine.medical_specialtyFilaminsCardiomyopathy030204 cardiovascular system & hematologySudden cardiac deathVentricular Dysfunction Left03 medical and health sciences0302 clinical medicineInterquartile rangeCardiac magnetic resonance imagingInternal medicinemedicineHumansConnectin030212 general & internal medicineHeart FailureEjection fractionmedicine.diagnostic_testbusiness.industryHazard ratioCorrectionStroke Volume[SDV.MHEP.EM]Life Sciences [q-bio]/Human health and pathology/Endocrinology and metabolismMiddle Agedmedicine.diseaseDefibrillators Implantable3. Good healthDeath Sudden CardiacCodon NonsenseHeart failureMutationCohortTachycardia VentricularCardiologyHeart TransplantationFemaleCardiology and Cardiovascular MedicinebusinessJAMA Cardiology
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Different expression of adrenoceptors and GRKs in the human myocardium depends on heart failure etiology and correlates to clinical variables.

2012

Downregulation of β1- adrenergic receptors (β1-ARs) and increased expression/function of G-protein-coupled receptor kinase 2 (GRK2) have been observed in human heart failure, but changes in expression of other ARs and GRKs have not been established. Another unresolved question is the incidence of these compensatory mechanisms depending on heart failure etiology and treatment. To analyze these questions, we quantified the mRNA/protein expressions of six ARs (α1A, α1B, α1D, β1, β2, and β3) and three GRKs (GRK2, GRK3, and GRK5) in left (LV) and right ventricle (RV) from four donors, 10 patients with ischemic cardiomyopathy (IC), 14 patients with dilated cardiomyopathy (DC), and 10 patients wit…

AdultCardiomyopathy DilatedMalemedicine.medical_specialtyGenotypePhysiologyCardiomyopathyMyocardial IschemiaVentricular Function LeftPhysiology (medical)Internal medicinemedicineHumansRNA MessengerHeart FailureAnalysis of VarianceEjection fractionIschemic cardiomyopathybiologybusiness.industryBeta adrenergic receptor kinaseMyocardiumDilated cardiomyopathyStroke VolumeStroke volumeMiddle Agedmedicine.diseaseG-Protein-Coupled Receptor KinasesReceptors AdrenergicEndocrinologymedicine.anatomical_structurePhenotypeVentricleSpainHeart failurebiology.proteinCardiologyLinear ModelsFemaleCardiology and Cardiovascular MedicinebusinessCardiomyopathiesAmerican journal of physiology. Heart and circulatory physiology
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Early carotid atherosclerosis and cardiac diastolic abnormalities in hypertensive subjects

2004

Despite the fact that it is known that hypertension may be associated to early atherosclerosis manifestations, few data are to date available on the relationship between early carotid abnormalities and left ventricular diastolic dysfunction. To address this issue, 142 hypertensive patients (64 females and 78 males) younger than 55 years, at the first diagnosis of mild-to-moderate essential hypertension (WHO/ISH criteria), were selected from a database consisting of 3541 subjects referred to ultrasound cardiovascular laboratory in the last 5 years. Carotid intima-media thickness (IMT) was detected by high-resolution vascular ultrasound and left ventricular structure and function by the use o…

AdultCarotid Artery DiseasesMalemedicine.medical_specialtyTime FactorsSettore MED/09 - Medicina Internaatherosclerosis carotid arteries diastoleDiastoleDoppler echocardiographyEssential hypertensionLeft ventricular hypertrophyVentricular Dysfunction LeftDiastoleInternal medicineInternal MedicineHumansMedicinecardiovascular diseasesUltrasonographymedicine.diagnostic_testbusiness.industryVascular diseaseMiddle Agedmedicine.diseaseSettore MED/11 - Malattie Dell'Apparato Cardiovascolaremedicine.anatomical_structureHypertensionCirculatory systemcardiovascular systemCardiologyFemalebusinessIsovolumic relaxation timeArtery
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Chronic Thromboembolic Pulmonary Hypertension: Pre- and Postoperative Assessment with Breath-hold MR Imaging Techniques

2004

To evaluate the potential of breath-hold magnetic resonance (MR) imaging techniques in morphologic and functional assessment of patients with chronic thromboembolic pulmonary hypertension (CTEPH) before and after surgery.Thirty-four patients with CTEPH were examined before and after pulmonary thromboendarterectomy (PTE). For morphologic assessment, contrast material-enhanced MR angiography was used; for assessment of hemodynamics, velocity-encoded gradient-echo sequences and cine gradient-echo sequences along the short axis of the heart were performed. Contrast-enhanced MR angiography was compared with selective digital subtraction angiography (DSA) for depiction of central thromboembolic m…

AdultGadolinium DTPAMalemedicine.medical_specialtyHypertension Pulmonarymedicine.medical_treatmentContrast MediaMagnetic Resonance Imaging CineEndarterectomyPulmonary ArterySensitivity and SpecificityVentricular Function LeftMagnetic resonance angiographyPostoperative ComplicationsHeart SeptumImage Processing Computer-AssistedmedicineHumansRadiology Nuclear Medicine and imagingProspective StudiesAgedThrombectomyEndarterectomymedicine.diagnostic_testPulmonary thromboendarterectomybusiness.industryHemodynamicsAngiography Digital SubtractionMagnetic resonance imagingDigital subtraction angiographyMiddle AgedImage Enhancementmedicine.diseaseMagnetic Resonance ImagingPulmonary hypertensionmedicine.anatomical_structureChronic DiseaseAngiographyVentricular Function RightFemaleRadiologyPulmonary EmbolismbusinessBlood Flow VelocityMagnetic Resonance AngiographyFollow-Up StudiesArteryRadiology
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