Search results for "Familial chylomicronaemia syndrome"

showing 4 items of 4 documents

Effectiveness and safety of lomitapide in a patient with familial chylomicronemia syndrome

2020

Background: Familial chylomicronemia syndrome (FCS) is characterized by severe fasting hypertriglyceridemia, abdominal pain, and recurrent acute pancreatitis. Available triglyceride-lowering drugs are insufficient to avoid pancreatitis. Therefore, there is a significant unmet medical need for effective triglyceride-lowering drugs for patients with FCS. Case report: We report the second case of a patient with FCS and recurrent pancreatitis treated with lomitapide. Lomitapide treatment resulted in a reduction of fasting TG levels from 2897 mg/dL (32.71 mmol/L) to an average of 954 mg/dL (10.77 mmol/L) on the 30 mg lomitapide equating to a 67% reduction from baseline. After 26 months of lomita…

Abdominal painPediatricsmedicine.medical_specialtyEndocrinology Diabetes and Metabolism030209 endocrinology & metabolismGastroenterology03 medical and health scienceschemistry.chemical_compound0302 clinical medicineEndocrinologyRecurrent pancreatitisInternal medicineFatty liverHumansMedicineProspective Studiesmedicine.diagnostic_testbusiness.industryFatty liverHypertriglyceridemiaFCSFamilial Chylomicronemiamedicine.diseaseLomitapideLomitapideAcute pancreatitisPancreatitischemistry030220 oncology & carcinogenesisLiver biopsyAcute DiseaseAcute pancreatitisPancreatitisBenzimidazolesHyperlipoproteinemia Type Ilipids (amino acids peptides and proteins)medicine.symptomCardiology and Cardiovascular MedicinebusinessFamilial chylomicronaemia syndromeAtherosclerosis
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Efficacy and safety of lomitapide in familial chylomicronaemia syndrome

2022

Familial chylomicronaemia syndrome (FCS) is a rare autosomal recessive disorder, resulting in elevated triglycerides (TGs), abdominal pain and pancreatitis. Treatment options are limited. Lomitapide, a microsomal triglyceride transfer protein inhibitor, is approved for the treatment of homozygous familial hypercholesterolaemia. Whether its therapeutic use may be extended to FCS remains unknown. The aim of this study was to evaluate the efficacy and safety of lomitapide in adult patients with FCS.The open-label, single-arm 'LOCHNES' study of lomitapide in FCS enrolled patients18 years with genetically confirmed FCS, elevated fasting TG ≥ 750 mg/dL and history of pancreatitis. Patients were a…

AdultPancreatitiSettore MED/09 - Medicina InternaTriglycerideBenzimidazoleLomitapideAbdominal PainPancreatitisHyperlipoproteinemia Type I.HumansBenzimidazolesHyperlipoproteinemia Type ICardiology and Cardiovascular MedicineTriglyceridesFamilial chylomicronaemia syndromeHuman
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Characterisation of patients with familial chylomicronaemia syndrome (FCS) and multifactorial chylomicronaemia syndrome (MCS): Establishment of an FC…

2018

Data presented in this article are supplementary material to our article entitled "Identification and diagnosis of patients with familial chylomicronaemia syndrome (FCS): expert panel recom mendations and proposal of an "FCS Score" (Moulin et al., 2018, in press). The data describe the genotypes of patients with familial chylomicronaemia syndrome (FCS) and multifactorial chylomicro naemia syndrome (MCS), from the validation and replication cohorts.

Settore MED/09 - Medicina InternadiagnosisMEDLINE030209 endocrinology & metabolism030204 cardiovascular system & hematologyBioinformaticslcsh:Computer applications to medicine. Medical informatics03 medical and health sciencesfamilial chylomicronaemia syndrome diagnostic score0302 clinical medicineDiagnòsticDiagnosisMalalties hereditàriesscoreMedicinelcsh:Science (General)Genetics Genomics and Molecular BiologyMultidisciplinarybusiness.industryfamilial chylomicronaemia syndrome (FCS) multifactorial chylomicronaemia syndrome (MCS) diagnosis scorefamilial chylomicronaemia syndrome (FCS)Rare diseaseslcsh:R858-859.7lipids (amino acids peptides and proteins)Malalties rareschylomicronaemia syndrome ; multifactorial chylomicronaemia syndromebusinessmultifactorial chylomicronaemia syndrome (MCS)Genetic diseaseslcsh:Q1-390Data in Brief
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Identification and diagnosis of patients with familial chylomicronaemia syndrome (FCS): Expert panel recommendations and proposal of an "FCS score".

2018

Familial chylomicronaemia syndrome (FCS) is a rare, inherited disorder characterised by impaired clearance of triglyceride (TG)-rich lipoproteins from plasma, leading to severe hypertriglyceridaemia (HTG) and a markedly increased risk of acute pancreatitis. It is due to the lack of lipoprotein lipase (LPL) function, resulting from recessive loss of function mutations in the genes coding LPL or its modulators. A large overlap in the phenotype between FCS and multifactorial chylomicronaemia syndrome (MCS) contributes to the inconsistency in how patients are diagnosed and managed worldwide, whereas the incidence of acute hypertriglyceridaemic pancreatitis is more frequent in FCS. A panel of Eu…

[SDV]Life Sciences [q-bio]Diagnosis toolpopulation030204 cardiovascular system & hematologyburdenapoa50302 clinical medicineLoss of Function MutationRisk FactorsChylomicrons030212 general & internal medicineAge of OnsetHypolipidemic AgentsBIOMEDICINA I ZDRAVSTVO. Kliničke medicinske znanosti. Interna medicina.Lipoprotein lipaseplasma triglycerideshyperlipoproteinemiaPrognosis3. Good healthUp-RegulationPhenotypeAcute pancreatitislipids (amino acids peptides and proteins)Hyperlipoproteinemia Type IAcute pancreatitis ; Familial chylomicronaemia syndrome ; Major hypertriglyceridaemia ; Multifactorial chylomicronaemiaCardiology and Cardiovascular MedicineFamilial chylomicronaemia syndromeAlgorithmsacute-pancreatitismedicine.medical_specialtyConsensushypertriglyceridemiaetiologyAcute pancreatitis; Familial chylomicronaemia syndrome; Major hypertriglyceridaemia; Multifactorial chylomicronaemia/Decision Support TechniquesDiagnosis Differential03 medical and health sciencesAcute pancreatitis; Familial chylomicronaemia syndrome; Major hypertriglyceridaemia; Multifactorial chylomicronaemia; Cardiology and Cardiovascular MedicinePredictive Value of TestsInternal medicinemedicineHumansGenetic Predisposition to DiseaseAcute pancreatitiBIOMEDICINE AND HEALTHCARE. Clinical Medical Sciences. Internal Medicine.GenotypingTriglyceridesPregnancyReceiver operating characteristicbusiness.industrysevereMultifactorial chylomicronaemiaReproducibility of Resultsmutationslipoprotein-lipase genemedicine.diseaseConfidence intervalAcute pancreatitisLipoprotein LipasePancreatitisCardiovascular System & CardiologyPancreatitisMajor hypertriglyceridaemiabusinessBiomarkersAtherosclerosis
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