Search results for "abnormal"

showing 10 items of 761 documents

Exclusion of PAX9 and MSX1 mutation in six families affected by tooth agenesis. A genetic study and literature review

2013

Objectives: In the present study, it is described the phenotypical analysis and the mutational screening, for genes PAX9 and MSX1, of six families affected by severe forms of tooth agenesis associated with other dental anomalies and systemic entities. Study Design: Six families affected by severe tooth agenesis associated with other dental anomalies and systemic entities were included. Oral exploration, radiological examination, medical antecedents consideration and mutational screening for PAX9 and MSX1 were carried out. Results: No mutations were discovered despite the fact that numerous teeth were missing. An important phenotypical variability was observed within the probands, not being …

ProbandMaleNonsense mutationOdontologíaGene mutationmedicine.disease_causeGene dosageAnodontiaGenetic transformationstomatognathic systemDental abnormalitiesmedicineMalalties hereditàriesMissense mutationHumansGeneral DentistryGenetic Association StudiesAnodontiaGeneticsMSX1 Transcription FactorMutationOral Medicine and Pathologybusiness.industryResearchmedicine.disease:CIENCIAS MÉDICAS [UNESCO]Ciencias de la saludFenotipstomatognathic diseasesPhenotypeOtorhinolaryngologyGenesUNESCO::CIENCIAS MÉDICASMutationSurgeryFemalePAX9 Transcription FactorbusinessMalformacions dentalsTransformació genèticaPAX9GensGenetic diseases
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Malformations in newborn: results based on 30,940 infants and fetuses from the Mainz congenital birth defect monitoring system (1990-1998).

2002

Prevalence rates of birth defects in the Federal Republic of Germany are informative to assess the general background risk of having a child with a birth defect. They provide basic figures to determine temporal and regional prevalence trends, to evaluate and initiate preventive measures and to initiate research projects. To avoid observer, definition and collection bias, active monitoring systems are required. Data collected in the active monitoring system of the Mainz Birth Defects Registry are presented. From 1990-1998, 30,940 live-births, stillbirths and abortions underwent standardized physical and sonographic examinations. Anamnestic data were collected from prenatal care records, mate…

ProbandMalePediatricsmedicine.medical_specialtyPolyhydramniosPrevalenceOligohydramniosPrenatal careCongenital AbnormalitiesCohort StudiesRisk FactorsGermanyPrevalenceMedicineHumansRegistriesPregnancybusiness.industryInfant NewbornObstetrics and GynecologyGestational ageGeneral Medicinemedicine.diseaseFemalebusinessCohort studyArchives of gynecology and obstetrics
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Excess of de novo variants in genes involved in chromatin remodelling in patients with marfanoid habitus and intellectual disability.

2020

PurposeMarfanoid habitus (MH) combined with intellectual disability (ID) (MHID) is a clinically and genetically heterogeneous presentation. The combination of array CGH and targeted sequencing of genes responsible for Marfan or Lujan–Fryns syndrome explain no more than 20% of subjects.MethodsTo further decipher the genetic basis of MHID, we performed exome sequencing on a combination of trio-based (33 subjects) or single probands (31 subjects), of which 61 were sporadic.ResultsWe identified eight genes with de novo variants (DNVs) in at least two unrelated individuals (ARID1B, ATP1A1, DLG4, EHMT1, NFIX, NSD1, NUP205 and ZEB2). Using simulation models, we showed that five genes (DLG4, NFIX, …

ProbandMale[SDV]Life Sciences [q-bio]intellectual deficiencyMESH: NFI Transcription Factorschromatin remodelingMarfan SyndromeCraniofacial AbnormalitiesMESH: ChildIntellectual disabilityMESH: Craniofacial AbnormalitiesMESH: Mental Retardation X-LinkedExomeChildde novo variantsGenetics (clinical)Exome sequencingGeneticsMESH: ExomeMESH: Middle AgedbiologyMESH: Genetic Predisposition to DiseaseMiddle AgedNFIXMESH: Young AdultFemaleAdultMESH: MutationAdolescentChromatin remodelingMESH: Intellectual DisabilityMESH: Marfan SyndromeEHMT1Young AdultMESH: Whole Exome SequencingIntellectual DisabilityExome SequencingGeneticsmedicineHumansGenetic Predisposition to Diseasemarfanoid habitusGeneMESH: Neurodevelopmental DisordersMESH: AdolescentMESH: HumansGenetic heterogeneityMESH: Chromatin Assembly and DisassemblyMESH: Histone-Lysine N-MethyltransferaseMESH: AdultHistone-Lysine N-Methyltransferasemedicine.diseaseChromatin Assembly and DisassemblyMESH: MaleNFI Transcription FactorsNeurodevelopmental DisordersMutationbiology.proteinMental Retardation X-LinkedMESH: FemaleJournal of medical genetics
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Contribution of molecular analyses in diagnosing Marfan syndrome and type I fibrillinopathies: an international study of 1009 probands.

2008

International audience; BACKGROUND: The diagnosis of Marfan syndrome (MFS) is usually initially based on clinical criteria according to the number of major and minor systems affected following international nosology. The number of FBN1 mutation carriers, at risk of aortic complications who would not be properly diagnosed based only on clinical grounds, is of growing importance owing to the increased availability of molecular screening. The aim of the study was to identify patients who should be considered for FBN1 mutation screening. METHODS: Our international series included 1009 probands with a known FBN1 mutation. Patients were classified as either fulfilling or not fulfilling "clinical"…

ProbandNosologyMarfan syndromeMalePediatricsSystemic diseaseMESH : International CooperationFibrillin-1International CooperationMESH : Aged[SDV.GEN] Life Sciences [q-bio]/GeneticsMarfan SyndromeMESH : ChildMESH: ChildEpidemiologyMESH : FemaleEctopia lentisChildGenetics (clinical)AortaAortic dissectionMESH: Aged0303 health sciences030305 genetics & heredityMicrofilament ProteinsMESH: AortaMESH : AdultConnective tissue disease3. Good healthFemaleMESH : Mutationmusculoskeletal diseasesAdultmedicine.medical_specialtycongenital hereditary and neonatal diseases and abnormalitiesMESH: MutationMESH : Microfilament ProteinsAdolescentMESH : MaleFibrillinsMESH: Marfan Syndrome03 medical and health sciencesMESH: Microfilament ProteinsMESH : AdolescentGeneticsmedicineHumans030304 developmental biologyAgedMESH: Adolescent[SDV.GEN]Life Sciences [q-bio]/GeneticsMESH : Marfan SyndromeMESH: Humansbusiness.industryMESH : HumansMESH : AortaMESH: Adultmedicine.diseaseMESH: MaleMESH: International CooperationMutation[ SDV.GEN ] Life Sciences [q-bio]/GeneticsbusinessMESH: FemaleJournal of medical genetics
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Toxic effects induced by vanadium on sea urchin embryos

2020

Vanadium, a naturally occurring element widely distributed in soil, water and air, has received considerable interest because its compounds are often used in different applications, from industry to medicine. While the possible medical use of vanadium compounds is promising, its potential harmful effects on living organisms are still unclear. Here, for the first time, we provide a toxicological profile induced by vanadium on Paracentrotus lividus sea urchin embryos, reporting an integrated and comparative analysis of the detected effects reflecting vanadium-toxicity. At the morphological level we found a dose-dependent induction of altered phenotypes and of skeletal malformations. At the mo…

Programmed cell deathEmbryo NonmammalianEnvironmental EngineeringHealth Toxicology and Mutagenesis0208 environmental biotechnologyVanadium-stressVanadiumchemistry.chemical_elementApoptosis02 engineering and technology010501 environmental sciences01 natural sciencesParacentrotus lividusDevelopmental abnormalityCellular stress responseHeat shock proteinAutophagyAnimalsHumansEnvironmental ChemistrySettore BIO/06 - Anatomia Comparata E Citologia0105 earth and related environmental sciencesHeat shock proteinsbiologyChemistryAutophagyPublic Health Environmental and Occupational HealthVanadiumGeneral MedicineGeneral Chemistrybiology.organism_classificationPollution020801 environmental engineeringCell biologyApoptosisParacentrotus lividus embryosToxicityParacentrotusChemosphere
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An organelle-specific protein landscape identifies novel diseases and molecular mechanisms.

2016

Cellular organelles provide opportunities to relate biological mechanisms to disease. Here we use affinity proteomics, genetics and cell biology to interrogate cilia: poorly understood organelles, where defects cause genetic diseases. Two hundred and seventeen tagged human ciliary proteins create a final landscape of 1,319 proteins, 4,905 interactions and 52 complexes. Reverse tagging, repetition of purifications and statistical analyses, produce a high-resolution network that reveals organelle-specific interactions and complexes not apparent in larger studies, and links vesicle transport, the cytoskeleton, signalling and ubiquitination to ciliary signalling and proteostasis. We observe sub…

Proteomics0301 basic medicineSystems AnalysisDNA Mutational Analysislnfectious Diseases and Global Health Radboud Institute for Molecular Life Sciences [Radboudumc 4]General Physics and AstronomyDatasets as Topicmethods [Chromatography Affinity]ProteomicsSensory disorders Donders Center for Medical Neuroscience [Radboudumc 12]Chromatography AffinityMass SpectrometryProtein Interaction Mappingtherapy [Ciliopathies]genetics [Ciliopathies]methods [Molecular Targeted Therapy]Molecular Targeted TherapyProtein Interaction MapsMultidisciplinaryCiliumChemistry (all)Qabnormalities [Spine]pathology [Ciliopathies]genetics [Muscle Hypotonia]therapy [Muscle Hypotonia]Metabolic Disorders Radboud Institute for Molecular Life Sciences [Radboudumc 6]metabolism [Proteins]isolation & purification [Proteins]physiology [Biological Transport]3. Good healthCell biologyVesicular transport proteinpathology [Dwarfism]metabolism [Cilia]Muscle Hypotoniaddc:500pathology [Muscle Hypotonia]pathology [Spine]genetics [Dwarfism]Rare cancers Radboud Institute for Health Sciences [Radboudumc 9]ScienceDwarfismExocystBiologyArticleGeneral Biochemistry Genetics and Molecular BiologyPhysics and Astronomy (all)03 medical and health sciencesIntraflagellar transportCiliogenesisOrganelleHumansCiliaBiochemistry Genetics and Molecular Biology (all)ProteinsBiological TransportGeneral Chemistrytherapy [Dwarfism]Fibroblastsgenetics [Proteins]CiliopathiesSpinemethods [Protein Interaction Mapping]Renal disorders Radboud Institute for Molecular Life Sciences [Radboudumc 11]030104 developmental biologyProteostasisHEK293 Cellsmethods [Proteomics]
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Early stimulation: psychomotor development of two girls with Aicardi syndrome.

1987

Summary The psychomotor development achieved by two girls with the Aicardi syndrome is described. Until now this syndrome has been considered to produce a total dissociation from the environment in those affected. The results of early intervention, although not spectacular, are sufficient to recommend early stimulation in these children.

Psychomotor learningPediatricsmedicine.medical_specialtyDissociation (neuropsychology)business.industryPublic Health Environmental and Occupational HealthInfantStimulationSyndromemedicine.diseaseLanguage DevelopmentDevelopmental psychologyAicardi syndromeBehavior TherapyIntervention (counseling)Pediatrics Perinatology and Child HealthDevelopmental and Educational PsychologymedicineHumansFemaleEye AbnormalitiesAgenesis of Corpus CallosumbusinessSpasms InfantilePsychomotor PerformanceChild: care, health and development
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The Speed of Incorporating Information into Prices

2013

Abstract To determine the speed of adjusting asset prices to the latest market information, investors usually resort to semi-strong form efficiency tests. Semi-strong form efficiency is based on the assumption that stock prices adjust rapidly as a result of new public information. The objective of the event study conducted in this paper was to examine whether new information is incorporated into the share price in a single price change after its public distribution. We analyzed the price behaviour of companies listed under Category I of the Bucharest Stock Exchange around events such interim result announcements between June and November 2012.

Public informationFinancial economicsGeneral EngineeringEvent studyEnergy Engineering and Power TechnologyShare priceinformationStock exchangeInterimPrice changeabnormal returnsEconomicsevent studyStock (geology)Procedia Economics and Finance
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Severe beak deformity in Melopsittacus undulatus caused by Knemidocoptes pilae

2014

An outbreak of knemidocoptic mange caused by Knemidocoptes pilae is described in caged Australian budgerigars (Melopsittacus undulatus) in a public park in L´Eliana (Valencia, Spain). Five individuals (5 out of 70) displayed severe malformation of the beak, with the rhamphotheca, rhinotheca, and gnathotheca affected and a slight lateral deviation of the jawbone, which did not, however, impede feeding or grooming.

Public parkGeneral VeterinaryKey words: Knemidocoptes pilaemangeMelopsittacus undulatusabnormal beak growthSpainLateral deviationMangeOutbreakAnatomyBiologyMelopsittacusmedicine.diseaseBeakDeformitymedicinemedicine.symptomKnemidocoptes pilae
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FVIII production by human lung microvascular endothelial cells

2006

While extrahepatic factor VIII (FVIII) synthesis suffices for hemostasis, the extrahepatic production sites are not well defined. We therefore investigated the ability of the human lungs to produce FVIII. Lungs from heart-beating donors who were declined for transplantation were perfused and ventilated in an isolated reperfusion model for 2 hours. A progressive accumulation of FVIII and von Willebrand factor (VWF) was recorded in the perfusion medium in 3 of 4 experiments. By contrast, factor V, fibrinogen, and immunoglobulin G (IgG) levels remained constant during the perfusion period, indicating that the accumulation of FVIII and VWF was not due to diffusion from the intercellular medium …

Pulmonary Circulationcongenital hereditary and neonatal diseases and abnormalitiesmedicine.medical_specialtyEndotheliumanimal diseasesImmunologyIn Vitro TechniquesFibrinogenBiochemistryImmunoglobulin GMicrocirculationVon Willebrand factorhemic and lymphatic diseasesInternal medicinevon Willebrand FactormedicineHumansLungFactor VIIILungbiologybusiness.industryMicrocirculationEndothelial CellsCell BiologyHematologyTransplantationKineticsEndocrinologymedicine.anatomical_structureHemostasisReperfusionImmunologybiology.proteinEndothelium Vascularbusinessmedicine.drugBlood
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