Search results for "Aminoaciduria"

showing 2 items of 2 documents

In Lysinuric Protein Intolerance system y+L activity is defective in monocytes and in GM-CSF-differentiated macrophages

2010

Abstract Background In the recessive aminoaciduria Lysinuric Protein Intolerance (LPI), mutations of SLC7A7/y+LAT1 impair system y+L transport activity for cationic amino acids. A severe complication of LPI is a form of Pulmonary Alveolar Proteinosis (PAP), in which alveolar spaces are filled with lipoproteinaceous material because of the impaired surfactant clearance by resident macrophages. The pathogenesis of LPI-associated PAP remains still obscure. The present study investigates for the first time the expression and function of y+LAT1 in monocytes and macrophages isolated from a patient affected by LPI-associated PAP. A comparison with mesenchymal cells from the same subject has been a…

AdultMaleCellular differentiationlcsh:MedicinePulmonary Alveolar ProteinosisBiologyMonocytesPathogenesisYoung AdultMacrophages AlveolarmedicineHumansGenetics(clinical)Pharmacology (medical)Amino Acid Metabolism Inborn ErrorsCells CulturedGenetics (clinical)Medicine(all)chemistry.chemical_classificationResearchFusion Regulatory Protein 1 Light ChainsLysinelcsh:RMesenchymal stem cellAmino Acid Transport System y+LGranulocyte-Macrophage Colony-Stimulating FactorCell DifferentiationGeneral Medicinemedicine.diseaseLysinuric protein intoleranceMolecular biologyAmino acidGranulocyte macrophage colony-stimulating factorchemistryAminoaciduriaImmunologyPulmonary alveolar proteinosismedicine.drugOrphanet Journal of Rare Diseases
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Association between M467T and 114 C--A variants within the SLC3A1 gene and some phenotypical traits in cystinuria patients from Spain.

2000

Cystinuria is an inherited metabolic disease characterized by an abnormal urinary excretion of cystine and dibasic amino acids. Formation of renal calculi, recurrent infections and renal failure are the main complications of this disease. The SLC3A1 gene, which codes for a dibasic amino acid transporter protein, is involved in the pathogenesis of cystinuria. We investigated the possible association between molecular variants (M467T, E483X, T216 M and 114 C--A) within the SLC3A1 gene and some phenotypical traits in a Spanish area. The study population consisted of 45 cystinuria patients, 42 cystinuria relatives and 81 healthy control subjects. Only the M467T mutation was found in chromosomes…

AdultMalemedicine.medical_specialtyGenotypeUrinary systemCystineBiologychemistry.chemical_compoundKidney CalculiSex FactorsMale Urogenital DiseasesInternal medicineGenotypeGeneticsmedicineHumansAlleleAmino AcidsChildAllele frequencyGenetics (clinical)AllelesCystinuriaMembrane GlycoproteinsPolymorphism GeneticMediterranean RegionAge FactorsCystinuriamedicine.diseaseFemale Urogenital DiseasesEndocrinologyPhenotypechemistrySpainAminoaciduriaMutationPopulation studyAmino Acid Transport Systems BasicRegression AnalysisFemaleCarrier ProteinsHuman genetics
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