Search results for " homozygous"

showing 10 items of 13 documents

Homozygous familial hypercholesterolemia in Italy: Clinical and molecular features

2020

Abstract Background and aims Homozygous familial hypercholesterolemia (HoFH) is a rare genetic disorder characterized by extremely elevated plasma levels of low density lipoprotein cholesterol (LDL-C) and high risk of premature atherosclerotic cardiovascular disease (ASCVD). HoFH is caused by pathogenic variants in several genes, such as LDLR, APOB and PCSK9, responsible for autosomal dominant hypercholesterolemia (ADH), and LDLRAP1 responsible for autosomal recessive hypercholesterolemia (ARH). Aim of this study was the review of the clinical and molecular features of patients with HoFH identified in Italy from 1989 to 2019. Methods Data were collected from lipid clinics and laboratories, …

Adult0301 basic medicinemedicine.medical_specialtyCandidate geneCandidate geneGenotype-phenotype correlationApolipoprotein BCandidate genes; Genotype-phenotype correlations; Homozygous familial hypercholesterolemia; Pathogenic variantsHomozygous familial hypercholesterolemiaGenotype-phenotype correlationsFamilial hypercholesterolemia030204 cardiovascular system & hematologyCompound heterozygosityCandidate genesHyperlipoproteinemia Type II03 medical and health sciences0302 clinical medicineInternal medicinemedicineHumansbiologybusiness.industryPCSK9HomozygoteGenetic disorderPathogenic variantsCandidate genes; Genotype-phenotype correlations; Homozygous familial hypercholesterolemia; Pathogenic variants;medicine.diseasePhenotype030104 developmental biologyEndocrinologyItalyReceptors LDLAutosomal Recessive HypercholesterolemiaMutationLDL receptorbiology.proteinlipids (amino acids peptides and proteins)Proprotein Convertase 9Cardiology and Cardiovascular Medicinebusiness
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Treating homozygous familial hypercholesterolemia in a real-world setting: Experiences with lomitapide

2015

Homozygous familial hypercholesterolaemia (HoFH) is a rare genetic disease characterised by markedly elevated plasma levels of low-density lipoprotein-cholesterol (LDL-C). Lomitapide is a microsomal triglyceride transfer protein (MTP) inhibitor approved as an adjunct to other lipid-lowering therapies (LLTs), with or without lipoprotein apheresis (LA), for the treatment of adult HoFH. Diet with <20% calories from fat is required. Due to a varying genetic and phenotypic profile of patients with HoFH, individual patients may respond to therapy differently; therefore examining individual cases in a 'real-world' setting provides valuable information on the effective day-to-day manag…

AdultMalemedicine.medical_specialtyCalorieSettore MED/09 - Medicina InternaEndocrinology Diabetes and MetabolismCase studyFamilial hypercholesterolemiaHomozygous familial hypercholesterolemiaFamilial hypercholesterolemiaDiseaseCompound heterozygosityHyperlipoproteinemia Type IIchemistry.chemical_compoundInternal medicineCase study; Familial hypercholesterolemia; Homozygous familial hypercholesterolemia; Lomitapide; Treatment; Cardiology and Cardiovascular Medicine; Endocrinology Diabetes and Metabolism; Internal Medicine; Nutrition and DieteticsInternal MedicinemedicineHumansAdverse effectNutrition and Dieteticsbusiness.industryAnticholesteremic AgentsHomozygoteCholesterol LDLMiddle Agedmedicine.diseaseLomitapideLomitapideTreatmentClinical trialEndocrinologychemistryBenzimidazolesFemaleSteatosisCardiology and Cardiovascular Medicinebusiness
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Extraction of Proteins From Leaves of Homozygous and Heterozygous Citrus for Two-Dimensional Electrophoresis Analysis.

2009

Citrus biothecnology proteomics citrus homozygous genotypes
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LDL apheresis in a homozygous familial hypercholesterolemic child aged 4.5.

1997

Preliminary experience with the efficacy and safety of dextran sulfate cellulose low-density lipoprotein (LDL) apheresis for the treatment of a 4.5-year-old girl with homozygous familial hypercholesterolemia and coronary artery disease is reported. The decrease of the most atherogenic apolipoprotein B-containing lipoproteins, low-density lipoprotein (LDL) and lipoprotein(a) (Lp [a]), were in the ranges of 63.1-68.7%, and 52.5-58.6%, respectively. The child tolerated LDL apheresis without any clinically significant complications. Therefore, she was submitted to a long-term program of treatment at intervals of 15 days. The experience suggests the possibility of an early beginning of extracorp…

Homozygous Familial Hypercholesterolemiamedicine.medical_specialtyApolipoprotein BBiomedical EngineeringMedicine (miscellaneous)BioengineeringCoronary Disease4.5 years-old girlFamilial hypercholesterolemiaBiomaterialsHyperlipoproteinemia Type IIchemistry.chemical_compoundInternal medicinemedicineCoronary Heart DiseaseHumansHyperlipoproteinemia Type IIbiologybusiness.industryCholesterolHomozygoteGeneral MedicineLipoprotein(a)Low Density Lipoprotein (LDL) apheresis; 4.5 years-old girl; Homozygous Familial Hypercholesterolemia; Coronary Heart Diseasemedicine.diseaseLipoproteins LDLApheresisEndocrinologyCholesterolLow Density Lipoprotein (LDL) apheresischemistryLDL apheresisChild Preschoolbiology.proteinBlood Component Removallipids (amino acids peptides and proteins)FemalebusinessLipoproteinLipoprotein(a)Artificial organs
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Management of chronic viral hepatitis in patients with thalassemia: recommendations from an international panel.

2010

AbstractChelation therapy with new drugs prevents cardiac damage and improves the survival of thalassemia patients. Liver diseases have emerged as a critical clinical issue. Chronic liver diseases play an important role in the prognosis of thalassemia patients because of the high frequency of viral infections and important role of the liver in regulating iron metabolism. Accurate assessment of liver iron overload is required to tailor iron chelation therapy. The diagnosis of hepatitis B virus– or hepatitis C virus–related chronic hepatitis is required to detect patients who have a high risk of developing liver complications and who may benefit by antiviral therapy. Moreover, clinical manage…

Liver Cirrhosisthalassemiamedicine.medical_specialtyCirrhosisC VIRUS-INFECTION; HOMOZYGOUS BETA-THALASSEMIA; TRANSFUSION-DEPENDENT THALASSEMIA; TERM-FOLLOW-UP; IRON OVERLOAD; LIVER-DISEASE; INTERFERON-ALPHA; RISK-FACTORS; INTRAFAMILIAL TRANSMISSION; HEPATOCELLULAR-CARCINOMAHepatitis C virusThalassemiaImmunologymedicine.disease_causeChronic liver diseaseAntiviral AgentsBiochemistryHOMOZYGOUS BETA-THALASSEMIALiver diseaseHepatitis B ChronicLIVER-DISEASEHEPATOCELLULAR-CARCINOMAmedicineTRANSFUSION-DEPENDENT THALASSEMIAIRON OVERLOADHumansIntensive care medicineTERM-FOLLOW-UPchronic viral hepatitis; thalassemia; managementbusiness.industryCell BiologyHematologyHepatitis CHepatitis C ChronicHepatitis Bmedicine.diseaseINTRAFAMILIAL TRANSMISSIONchronic viral hepatitisImmunologyRISK-FACTORSINTERFERON-ALPHAViral hepatitisbusinessC VIRUS-INFECTIONmanagement
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Individual analysis of patients with HoFH participating in a phase 3 trial with lomitapide: The Italian cohort

2015

Abstract Background and aims The efficacy and safety of lomitapide as adjunct treatment for adults with homozygous familial hypercholesterolaemia (HoFH) have been confirmed in a phase 3 trial. Given the small number of patients (N = 29), and variations in patient characteristics, examining individual cases provides additional details regarding patient management with lomitapide. Here, we examine the details of the Italian patient cohort in the phase 3 trial. Methods and results The methodology of the multinational, single-arm, open-label, 78-week, dose-escalation, phase 3 trial has been previously reported. The current report details the Italian cohort of six patients (three males, three fe…

MalePediatricsTime FactorsSettore MED/09 - Medicina InternaEndocrinology Diabetes and MetabolismHoFHMedicine (miscellaneous)030204 cardiovascular system & hematologychemistry.chemical_compound0302 clinical medicineEndocrinologyReceptorsNutrition and DieteticMedicine030212 general & internal medicineFamilial hypercholesterolaemia; FH; HoFH; Homozygous familial hypercholesterolaemia; Lomitapide; Therapy; Medicine (miscellaneous); Nutrition and Dietetics; Endocrinology Diabetes and Metabolism; Cardiology and Cardiovascular MedicineFH; Familial hypercholesterolaemia; HoFH; Homozygous familial hypercholesterolaemia; Lomitapide; Therapy; Adolescent; Adult; Anticholesteremic Agents; Benzimidazoles; Biomarkers; Cholesterol LDL; Female; Genetic Predisposition to Disease; Humans; Hyperlipoproteinemia Type II; Italy; Male; Middle Aged; Phenotype; Receptors LDL; Time Factors; Treatment Outcome; Young Adult; Heterozygote; MutationNutrition and DieteticsAnticholesteremic AgentsMiddle AgedPatient managementDiabetes and MetabolismCholesterolPhenotypeTreatment OutcomeTolerabilityItalyCohortPopulation studyFemaleFamilial hypercholesterolaemiaCardiology and Cardiovascular MedicineAdultmedicine.medical_specialtyHeterozygoteAdolescentSocio-culturaleFHLDLHyperlipoproteinemia Type II03 medical and health sciencesIndividual analysisYoung AdultHomozygous familial hypercholesterolaemiaHumansGenetic Predisposition to DiseaseAdverse effectbusiness.industryCholesterol LDLLomitapideLomitapideClinical trialchemistryReceptors LDLMutationBenzimidazolesTherapybusinessBiomarkers
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Efficacy of Lomitapide in the Treatment of Familial Homozygous Hypercholesterolemia: Results of a Real-World Clinical Experience in Italy

2017

Homozygous familial hypercholesterolaemia (HoFH) is a rare form of inherited dyslipidemia resistant to conventional cholesterol-lowering medications so that lipoprotein apheresis (LA) is usually required. Lomitapide has been approved for the treatment of HoFH. The aim of this study was to evaluate the benefits of lomitapide in HoFH patients followed with the usual clinical care. Homozygous familial hypercholesterolaemia (HoFH) is a rare form of inherited dyslipidemia resistant to conventional cholesterol-lowering medications so that lipoprotein apheresis (LA) is usually required. Lomitapide has been approved for the treatment of HoFH. The aim of this study was to evaluate the benefits of lo…

MaleSettore MED/09 - Medicina InternaHyperlipidemia Familial Combined030204 cardiovascular system & hematologyPharmacologyBenzimidazolecholesterol-lowering effect; clinical practice; genetics; lomitapide; severe hypercholesterolemia; medicine (all); pharmacology (medical)cholesterol-lowering effectchemistry.chemical_compound0302 clinical medicineRetrospective StudieAnticholesteremic Agentgenetics030212 general & internal medicineAged 80 and overAnticholesteremic AgentsHomozygoteGeneral MedicineMiddle Agedclinical practiceSafety profileItalylipids (amino acids peptides and proteins)FemaleHumanAdultmedicine.medical_specialtySocio-culturaleLiver ultrasoundLDLRAP1 geneHyperlipoproteinemia Type II03 medical and health sciencesGeneticInternal medicinemedicineHumansLiver damagemedicine (all)Familial homozygous hypercholesterolemiaAgedRetrospective Studieslomitapidebusiness.industrysevere hypercholesterolemiamedicine.diseaseRheumatologyLomitapidepharmacology (medical)chemistryBenzimidazolesbusinessDyslipidemia
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Efficacy and safety of a microsomal triglyceride transfer protein inhibitor in patients with homozygous familial hypercholesterolaemia: a single-arm,…

2013

Summary Background Patients with homozygous familial hypercholesterolaemia respond inadequately to existing drugs. We aimed to assess the efficacy and safety of the microsomal triglyceride transfer protein inhibitor lomitapide in adults with this disease. Methods We did a single-arm, open-label, phase 3 study of lomitapide for treatment of patients with homozygous familial hypercholesterolemia. Current lipid lowering therapy was maintained from 6 weeks before baseline through to at least week 26. Lomitapide dose was escalated on the basis of safety and tolerability from 5 mg to a maximum of 60 mg a day. The primary endpoint was mean percent change in levels of LDL cholesterol from baseline …

Malemedicine.medical_specialtySettore MED/09 - Medicina InternaMipomersenPhases of clinical researchSocio-culturaleFamilial hypercholesterolemialdl-apheresismtp inhibitorBenzimidazoleMicrosomal triglyceride transfer proteinHyperlipoproteinemia Type IIchemistry.chemical_compoundlipid lowering therapyInternal medicineClinical endpointMedicinelomitapidebiologybusiness.industryCholesterolMedicine (all)Homozygotelomitapide; ldl-apheresis; lipid lowering therapy; homozygous familial hypercholesterolemia; mtp inhibitorGeneral MedicineCholesterol LDLhomozygous familial hypercholesterolemiamedicine.diseaseLomitapideEndocrinologyTolerabilitychemistrybiology.proteinFemalebusinessCarrier ProteinHuman
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New Frontiers in the Treatment of Homozygous Familial Hypercholesterolemia.

2021

: Homozygous familial hypercholesterolemia (HoFH) is a rare genetic disorder. The most common cause is a mutation in both alleles of the gene encoding for the low-density lipoprotein (LDL) receptor, although other causative mutations have been identified. Complications of atherosclerotic cardiovascular disease are common in these patients; therefore, reducing the elevated LDL-cholesterol burden is critical in their management. Conventionally, this is achieved by patients initiating lipid-lowering therapy, but this can present challenges in clinical practice. Fortunately, novel therapeutic strategies have enabled promising innovations in HoFH treatment. This review highlights recent and ongo…

Settore MED/09 - Medicina InternaGenetic enhancementHomozygous familial hypercholesterolemiaFamilial hypercholesterolemiaInclisiranBioinformaticsmedicine.disease_causeBenzimidazolePCSK9Hyperlipoproteinemia Type IIchemistry.chemical_compoundGene therapyAnticholesteremic AgentmedicineAngiopoietin-like 3HumansLow-density lipoprotein cholesterolAlleleAngiopoietin-like 3; Gene therapy; Gene-editing; Homozygous familial hypercholesterolemia; Inclisiran; Lomitapide; Low-density lipoprotein cholesterol; PCSK9MutationGene-editingAtherosclerotic cardiovascular diseasebusiness.industryPCSK9Anticholesteremic AgentsHomozygoteGenetic disorderGeneral MedicineCholesterol LDLmedicine.diseaseLomitapideLomitapidechemistrylipids (amino acids peptides and proteins)BenzimidazolesCardiology and Cardiovascular MedicinebusinessHumanHeart failure clinics
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A novel therapeutic strategy to cure the Homozygous Familial Hypercholesterolemia with residual LDL receptor activity

2020

Settore MED/09 - Medicina InternaLDLR Homozygous Familial Hypercholesterolemia PCSK9 IDOL
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