Search results for "Membrane Transport"

showing 10 items of 215 documents

Recessive multiple epiphyseal dysplasia (rMED): phenotype delineation in eighteen homozygotes for DTDST mutation R279W.

2003

Multiple epiphyseal dysplasia (MED) is a generalised skeletal dysplasia that although relatively mild is associated with significant morbidity. Joint pain, joint deformity, waddling gait, and short stature are the main clinical signs and symptoms. In the past, the disorder was subdivided into the milder Ribbing type, usually with flattened epiphyses,1 and the more severe Fairbank type with round epiphyses,2 but many cases were not classifiable as clearly either type.3 MED can be caused by mutations in at least six separate genes: COMP ,4–7 collagen IX ( COL9A1 , COL9A2 , and COL9A3 ),8–13 matrilin 3 ( MATN3 ),15 and the sulphate transporter, DTDST ( DTDST/SLC26A2 ). We have previously repor…

AdultMalePathologymedicine.medical_specialtyAdolescentAnion Transport ProteinsGenes RecessiveBiologySLC26A2ArginineOsteochondrodysplasiasShort statureMultiple epiphyseal dysplasiaGeneticsmedicineHumansChildGenetics (clinical)GeneticsAchondrogenesisSulfatesPoint mutationHomozygoteTryptophanChromosome MappingMembrane Transport ProteinsBiological TransportMiddle Agedmedicine.diseasePhenotypeGenetic defects of metabolism [UMCN 5.1]Amino Acid SubstitutionDysplasiaSulfate TransportersMutation (genetic algorithm)MutationMutation testingbiology.proteinFemalemedicine.symptomCarrier ProteinsLetter to JMGJournal of medical genetics
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Mutations in ARL2BP, Encoding ADP-Ribosylation-Factor-Like 2 Binding Protein, Cause Autosomal-Recessive Retinitis Pigmentosa

2013

Retinitis pigmentosa (RP) is a genetically heterogeneous retinal degeneration characterized by photoreceptor death, which results in visual failure. Here, we used a combination of homozygosity mapping and exome sequencing to identify mutations in ARL2BP, which encodes an effector protein of the small GTPases ARL2 and ARL3, as causative for autosomal-recessive RP (RP66). In a family affected by RP and situs inversus, a homozygous, splice-acceptor mutation, c.101−1G>C, which alters pre-mRNA splicing of ARLBP2 in blood RNA, was identified. In another family, a homozygous c.134T>G (p.Met45Arg) mutation was identified. In the mouse retina, ARL2BP localized to the basal body and cilium-associated…

AdultMaleRetinal degenerationCentrioleMolecular Sequence DataGenes RecessiveBiologymedicine.disease_causeMice03 medical and health sciences0302 clinical medicineBardet–Biedl syndromeGTP-Binding ProteinsReportRetinitis pigmentosaGeneticsmedicineAnimalsHumansBasal bodyGenetics(clinical)Photoreceptor CellsGenetics (clinical)030304 developmental biologyPrimary ciliary dyskinesiaGenetics0303 health sciencesMutationBase SequenceADP-Ribosylation FactorsCiliumHomozygoteMembrane Transport ProteinsEpithelial Cellsmedicine.diseasePedigreeCell biologyMutationFemalesense organsCarrier ProteinsRetinitis Pigmentosa030217 neurology & neurosurgeryProtein BindingTranscription FactorsThe American Journal of Human Genetics
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Adult attachment and gene polymorphisms of the dopamine D4 receptor and serotonin transporter (5-HTT).

2010

Recently, the Dopamine D4 Receptor Gene (DRD4) and the Serotonin Transporter Gene (5-HTT) have been found to be candidate genes for infant attachment disorganization. The present study aimed to explore the relationship of these genes to adult attachment representations. The Adult Attachment Interview was used to assess attachment representations in 167 German adults. DNA from buccal cells was genotyped for the DRD4 VNTR Exon III and 5-HTT LPR polymorphisms with respect to the presence of the 7repeat allele and the short allele, respectively. DRD4 7repeat allele carriers were significantly more likely to be securely attached than those without 7repeat but only for subjects with unloving care…

AdultMalemedicine.medical_specialtyCandidate geneGenotypePsychometricsStatistics as TopicExonDopamineRisk Factorsmental disordersInterview PsychologicalDevelopmental and Educational PsychologymedicineAttachment theoryHumansAllelePsychiatryGeneSerotonin transporterRetrospective StudiesGeneticsSerotonin Plasma Membrane Transport ProteinsPolymorphism GeneticbiologyReceptors Dopamine D4Middle AgedObject AttachmentPsychiatry and Mental healthCross-Sectional StudiesPhenotypebiology.proteinFemalePsychologyAttachment measuresmedicine.drugAttachmenthuman development
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Norepinephrine transporter gene polymorphism is not associated with susceptibility to alcohol dependence

2002

Abnormalities in monoamine neurotransmission have been implicated in the pathogenesis of alcoholism, mood disorders and schizophrenia. Murine norepinephrine transporter gene (NET) has been mapped to a region on chromosome 8 where a quantitative trait locus for ethanol sensitivity. Therefore we tested whether norepinephrine transporter (NET) gene variants confer susceptibility to either alcohol dependence or severe alcohol withdrawal symptoms. There is a highly polymorphic silent G1287A mutation in the NET gene. In our study 157 alcoholics and 185 healthy unrelated matched control subjects were analyzed for a silent G1287A mutation. No significant differences in allele and genotype distribut…

AdultMalemedicine.medical_specialtyGenotypeDNA Mutational AnalysisMolecular Sequence DataAlcohol Withdrawal DeliriumGene FrequencyPolymorphism (computer science)Internal medicineGenotypemedicineHumansGenetic Predisposition to DiseaseRNA MessengerAlleleAllelesBiological PsychiatryGeneticsNorepinephrine Plasma Membrane Transport ProteinsPolymorphism GeneticSymportersbiologybusiness.industryAlcohol dependenceExonsMiddle Agedmedicine.diseaseAlcoholismPsychiatry and Mental healthMonoamine neurotransmitterEndocrinologyMood disordersNorepinephrine transporterbiology.proteinFemaleGene polymorphismbusinessPolymorphism Restriction Fragment LengthPsychiatry Research
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Evidence for the importance of the human dopamine transporter gene for withdrawal symptomatology of alcoholics in a German population

2002

Two new polymorphisms in the 3' untranslated region (3'UTR) of the dopamine transporter (DAT1) gene, adjacent to the known variable number of tandem repeats (VNTR) polymorphism, have been investigated in the present population-based association study including 351 alcoholics and 336 controls. The DraI restriction site was not polymorphic in our population. The G2319A polymorphism was not significantly different with respect to genotype or allele distribution between alcoholics and controls. Subsequently, in individuals with VNTR homozygosity for the ten repeat allele, we found a higher prevalence of A/A homozygosity in patients with seizure history (P = 0.001, odds ratio (OR) = 7.913), with…

AdultMalemedicine.medical_specialtyGenotypeMolecular Sequence DataPopulationNerve Tissue ProteinsGene FrequencyPolymorphism (computer science)GermanyInternal medicineGenotypeOdds RatiomedicineHumansAlleleeducationDopamine transporterGeneticsDopamine Plasma Membrane Transport Proteinseducation.field_of_studyChi-Square DistributionMembrane GlycoproteinsPolymorphism GeneticbiologyGeneral NeuroscienceMembrane Transport ProteinsOdds ratioMiddle AgedSubstance Withdrawal SyndromeAlcoholismRestriction siteVariable number tandem repeatEndocrinologybiology.proteinFemaleNeuroscience Letters
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Analysis of thiamine transporter genes in sporadic beriberi

2014

Abstract Objective Thiamine or vitamin B 1 deficiency diminishes thiamine-dependent enzymatic activity, alters mitochondrial function, impairs oxidative metabolism, and causes selective neuronal death. We analyzed for the first time, the role of all known mutations within three specific thiamine carrier genes, SLC19 A2, SLC19 A3 , and SLC25 A19 , in a patient with atrophic beriberi, a multiorgan nutritional disease caused by thiamine deficiency. Methods A 44-year-old male alcoholic patient from Morocco developed massive bilateral leg edema, a subacute sensorimotor neuropathy, and incontinence. Despite normal vitamin B 1 serum levels, his clinical picture was rapidly reverted by high-dose in…

AdultMalemedicine.medical_specialtySLC19 A- SLC25 A19SLC19 AEndocrinology Diabetes and MetabolismGene mutationBeriberimedicine.disease_causeMitochondrial Membrane Transport Proteinslaw.inventionBeriberilawInternal medicineGenotypemedicineThiamine transporterObjective: Thiamine or vitamin B1 deficiency diminishes thiamine-dependent enzymatic activity alters mitochondrial function impairs oxidative metabolism and causes selective neuronal death. We analyzed for the first time the role of all known mutations within three specific thiamine carrier genes SLC19 A2 SLC19 A3 and SLC25 A19 in a patient with atrophic beriberi a multiorgan nutritional disease caused by thiamine deficiency. Methods: A 44-year-old male alcoholic patient from Morocco developed massive bilateral leg edema a subacute sensorimotor neuropathy and incontinence. Despite normal vitamin B1 serum levels his clinical picture was rapidly reverted by high-dose intramuscular thiamine treatment suggesting a possible genetic resistance. We used polymerase chain reaction followed by amplicon sequencing to study all the known thiamine-related gene mutations identified within the Human Gene Mutation Database. Results: Thirty-seven mutations were tested: 29 in SLC19 A2 6 in SLC19 A3 and 2 in SLC25 A19. Mutational analyses showed a wild-type genotype for all sequences investigated. Conclusion: This is the first genetic study in beriberi disease. We did not detect any known mutation in any of the three genes in a sporadic dry beriberi patient. We cannot exclude a role for other known or unknown mutations in the same genes or in other thiamine-associated genes in the occurrence of this nutritional neuropathy.HumansThiamineGenePolymerase chain reactionGeneticsMutationNutrition and DieteticsbiologyMembrane Transport ProteinsThiamine Deficiencymedicine.diseaseAlcoholismEndocrinologyMutationbiology.proteinThiamineMutations
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Interaction between gene variants of the serotonin transporter promoter region (5-HTTLPR) and catecholO-methyltransferase (COMT) in borderline person…

2008

Borderline personality disorder (BPD) is characterized by a heterogeneous symptomatology with instability in impulse control, interpersonal relationships and self-image. BPD patients display repeated self-injury, chronic suicidal tendencies and emotional dysregulation, mainly dysregulation of negative affect. In its etiology, genetic and environmental factors have been suggested. Recently, an investigation in male healthy volunteers found gene–gene effects of the catechol-O-methyl-transferase (COMT) low-activity (Met158) and the low-expression allele of the deletion/insertion (short/long or S/L, respectively) polymorphism in the serotonin transporter-linked promoter region (5-HTTLPR) on the…

AdultMalemedicine.medical_specialtySingle-nucleotide polymorphismCatechol O-MethyltransferasePolymorphism Single Nucleotidebehavioral disciplines and activitiesCellular and Molecular NeuroscienceGene FrequencyGene interactionBorderline Personality DisorderInternal medicinemental disordersGenotypemedicineHumansAllelePromoter Regions GeneticBorderline personality disorderAllelesGenetics (clinical)Serotonin transporterSerotonin Plasma Membrane Transport ProteinsGeneticsCatechol-O-methyl transferasebiologybusiness.industryMiddle Agedmedicine.diseasePsychiatry and Mental healthLogistic ModelsEndocrinology5-HTTLPRbiology.proteinFemalebusinessAmerican Journal of Medical Genetics Part B: Neuropsychiatric Genetics
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Intrafamilial variability of the deafness and goiter phenotype in Pendred syndrome caused by a T416P mutation in the SLC26A4 gene.

2004

AbstractPendred syndrome (PS) is the most common cause of syndromic deafness, accounting for more than 5% of all autosomal-recessive hearing loss cases. It is characterized by bilateral sensorineural hearing loss and by goiter with or without hypothyroidism. Mutations in the SLC26A4 gene cause both classical PS and deafness associated with an enlarged vestibular aqueduct without goiter.To investigate a possible genotype-phenotype correlation in PS, we performed a detailed clinical and genetic study in three adult German sibs with typical PS caused by a common homozygous SLC26A4 mutation, T416P. An audiological long-term follow-up of 23 yr showed that the mutation T416P is associated with a …

AdultMalemedicine.medical_specialtyVestibular aqueductGoiterAdolescentHearing lossEndocrinology Diabetes and Metabolismmedicine.medical_treatmentHearing Loss SensorineuralClinical BiochemistryThyroid GlandDeafnessBiochemistryConnexinsEndocrinologyInternal medicineotorhinolaryngologic diseasesmedicineHumansChildPendred syndromebusiness.industryGoiterBiochemistry (medical)ThyroidThyroidectomyMembrane Transport ProteinsSyndromemedicine.diseaseConnexin 26Endocrinologymedicine.anatomical_structurePhenotypeSulfate TransportersChild PreschoolMutationSensorineural hearing lossFemalemedicine.symptombusinessEnlarged vestibular aqueductThe Journal of clinical endocrinology and metabolism
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Genetic polymorphisms of serotonin transporter and receptor 1A could influence success during embryo implantation and maintenance of pregnancy

2012

Objective To explore whether serotonin-related gene polymorphisms influence clinical outcomes of IVF treatment in recipients using donated oocytes. Design Nested case-control study. Setting University-affiliated infertility clinic. Patient(s) Two hundred forty-five women undergoing IVF treatment with donated oocytes. Intervention(s) None. Main Outcome Measure(s) Genotype and haplotype analysis of the serotonin transporter-linked polymorphic region (5-HTTLPR), rs1800532, rs6295, rs6313, and rs3813929, between recipients grouped according to the results of the oocyte donation for IVF treatment. Result(s) No differences were found between genotype distribution of the tryptophan hydroxylase 1, …

Adultmedicine.medical_specialtymedicine.medical_treatmentEarly Pregnancy LossFertilization in VitroPolymorphism Single NucleotideAndrologyGene FrequencyPregnancyRisk FactorsInternal medicineGenotypeOdds RatiomedicineHumansEmbryo ImplantationSerotonin transporterSerotonin Plasma Membrane Transport ProteinsAnalysis of VariancePregnancyChi-Square DistributionIn vitro fertilisationOocyte DonationbiologyHaplotypeObstetrics and GynecologyEmbryo Transfermedicine.diseaseEmbryo transferAbortion SpontaneousPregnancy rateLogistic ModelsPhenotypeTreatment OutcomeEndocrinologyHaplotypesReproductive MedicineCase-Control StudiesReceptor Serotonin 5-HT1Abiology.proteinFemaleFertility and Sterility
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CB1 cannabinoid receptor-mediated aggressive behavior

2013

This study examined the role of cannabinoid CB1 receptors (CB1r) in aggressive behavior. Social encounters took place in grouped and isolated mice lacking CB1r (CB1KO) and in wild-type (WT) littermates. Cognitive impulsivity was evaluated in the delayed reinforcement task (DRT). Gene expression analyses of monoaminooxidase-A (MAO-A), catechol-o-methyl-transferase (COMT), 5-hydroxytriptamine transporter (5-HTT) and 5-HT1B serotonergic receptor (5HT1Br) in the median and dorsal raphe nuclei (MnR and DR, respectively) and in the amygdala (AMY) were performed by real time-PCR. Double immunohistochemistry studies evaluated COMT and CB1r co-localization in the raphe nuclei and in the cortical (AC…

AgonistMalemedicine.medical_specialtyCannabinoid receptorTime Factorsmedicine.drug_classmedicine.medical_treatmentPoison controlArachidonic AcidsSerotonergicCatechol O-MethyltransferaseAmygdalaCellular and Molecular NeuroscienceMiceDorsal raphe nucleusReceptor Cannabinoid CB1Internal medicinemedicineAnimalsInterpersonal RelationsMonoamine OxidasePharmacologyCannabinoid Receptor AgonistsMice KnockoutSerotonin Plasma Membrane Transport ProteinsAmygdalaSurgeryAggressionmedicine.anatomical_structureEndocrinologynervous systemGene Expression RegulationImpulsive BehaviorReceptor Serotonin 5-HT1BConditioning OperantRaphe NucleiCannabinoidRaphe nucleiPsychologyReinforcement Psychology
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