Search results for "chylomicronemia"

showing 5 items of 5 documents

The role of registries in rare genetic lipid disorders: Review and introduction of the first global registry in lipoprotein lipase deficiency

2017

International audience; A good understanding of the natural history of rare genetic lipid disorders is a pre-requisite for successful patient management. Disease registries have been helpful in this regard. Lipoprotein Lipase Deficiency (LPLD) is a rare, autosomal-recessive lipid disorder characterized by severe hypertriglyceridemia and a very high risk for recurrent acute pancreatitis, however, only limited data are available on its natural course. Alipogene tiparvovec (Glybera (R)) is the first gene therapy to receive Marketing Authorization in the European Union; GENIALL (GENetherapy In the MAnagement of Lipoprotein Lipase Deficiency), a 15-year registry focusing on LPLD was launched in …

0301 basic medicinePediatricsPathologySettore MED/09 - Medicina Interna[SDV]Life Sciences [q-bio]Familial hypercholesterolemiaDisease030204 cardiovascular system & hematologyGeneTHERAPY0302 clinical medicineFamilialRisk FactorsHyperchylomicronemiaAlipogene tiparvovecRegistriesFAMILIAL HYPERCHOLESTEROLEMIAmedia_commonHypertriglyceridemiaPrognosis3. Good healthNatural historySystematic reviewPhenotypeDISEASESSAFETYHyperlipoproteinemia Type ICardiology and Cardiovascular Medicinemedicine.medical_specialtyAPHERESISRegistryFamilial chylomicronemia syndromeGENIALLLysosomal acid lipase deficiencyLipid Metabolism Inborn Errors03 medical and health sciencesLipoprotein lipase deficiencyRare DiseasesGene therapychylomicronemia syndromemedicinemedia_common.cataloged_instanceHumansGenetic Predisposition to DiseaseEuropean unionLipoprotein lipase deficiency (LPLD)business.industryALIPOGENE TIPARVOVEC AAV1-LPLS447Xmedicine.diseaseAlipogene tiparvovecLipoprotein Lipase030104 developmental biologyOrphan diseasebusiness
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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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Diagnostic algorithm for familial chylomicronemia syndrome

2016

International audience; Background: Familial chylomicronemia syndrome (FCS) is a rare genetic disease that leads to severe hypertriglyceridemia often associated with recurrent episodes of pancreatitis. The recognition and correct diagnosis of the disease is challenging due to its rarity, and to the lack of specificity of signs and symptoms. Lipid experts, endocrinologists, gastroenterologists, pancreatologists, and general practitioners may encounter patients who potentially have FCS. Therefore, cooperation between experts and improved knowledge of FCS is essential in improving the diagnosis. Currently, a consensus on best practice for the diagnosis of FCS is lacking. Methods: Aiming to def…

Chylomicrons; Familial chylomicronemia syndrome; Hyperlipoproteinemia; Lipoprotein lipase deficiency; Pancreatitis; Biomarkers; Genetic Markers; Genetic Predisposition to Disease; Humans; Hyperlipoproteinemia Type I; Lipids; Lipoprotein Lipase; Phenotype; Practice Guidelines as Topic; Predictive Value of Tests; Prognosis; Algorithms; Critical Pathways; DNA Mutational Analysis; Decision Support Techniques; Mutation; Internal Medicine; Cardiology and Cardiovascular MedicineSettore MED/09 - Medicina InternaACUTE-PANCREATITIS[SDV]Life Sciences [q-bio]DNA Mutational AnalysisPredictive Value of TestDisease030204 cardiovascular system & hematologyVARIANTSDecision Support Technique0302 clinical medicineDOMAINGenetic MarkerBINDINGChylomicronsHYPERTRIGLYCERIDEMICMedicine030212 general & internal medicinePANCREATITISLipoprotein lipase deficiencyGeneral MedicineFamilial ChylomicronemiaLipidPrognosisLipids3. Good healthAlgorithmDEFICIENCYPhenotypeCritical PathwayPractice Guidelines as TopicCritical PathwaysHyperlipoproteinemia Type Ilipids (amino acids peptides and proteins)Cardiology and Cardiovascular MedicineAlgorithmAlgorithmsHumanGenetic MarkersSevere hypertriglyceridemiaFamilial chylomicronemia syndromePrognosiSigns and symptomsLIPOPROTEIN-LIPASEHyperlipoproteinemiaCLASSIFICATIONDecision Support TechniquesSecondary careChylomicronDNA Mutational Analysi03 medical and health sciencesPredictive Value of TestsInternal MedicineMANAGEMENTHumansGenetic Predisposition to DiseasePancreatitibusiness.industryBiomarkerLipoprotein LipaseMutationbusinessBiomarkers
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Spectrum of mutations of the LPL gene identified in Italy in patients with severe hypertriglyceridemia.

2015

Background: Monogenic hypertriglyceridemia (HTG) may result from mutations in some genes which impair the intravascular lipolysis of triglyceride (TG)-rich lipoproteins mediated by the enzyme Lipoprotein lipase (LPL). Mutations in the LPL gene are the most frequent cause of monogenic HTG (familial chylomicronemia) with recessive transmission. Methods: The LPL gene was resequenced in 149 patients with severe HTG (TG>10mmol/L) and 106 patients with moderate HTG (TG>4.5 and <10mmol/L) referred to tertiary Lipid Clinics in Italy. Results: In the group of severe HTG, 26 patients (17.4%) were homozygotes, 9 patients (6%) were compound heterozygotes and 15 patients (10%) were simple heter…

MaleSettore MED/09 - Medicina InternaDNA Mutational AnalysisFamilial chylomicronemia; Gene variants; Lipoprotein lipase; Pancreatitis; Primary hypertriglyceridemiaCompound heterozygositymedicine.disease_causeSeverity of Illness IndexPrimary hypertriglyceridemiaTertiary Care Centerschemistry.chemical_compoundGene FrequencyFamilial chylomicronemia; Gene variants; Lipoprotein lipase; Pancreatitis; Primary hypertriglyceridemia; Cardiology and Cardiovascular MedicineChildLipoprotein lipaseMutationHomozygoteMiddle AgedPhenotypeItalyChild PreschoolFemaleHyperlipoproteinemia Type ICardiology and Cardiovascular MedicineFamilial chylomicronemiaAdultmedicine.medical_specialtyGene variantHeterozygoteAdolescentBiologyGene variantsYoung AdultInternal medicinemedicineLipolysisHumansGenetic Predisposition to DiseaseTriglyceridesAgedPancreatitiTriglycerideHypertriglyceridemiaInfantHeterozygote advantageLipoprotein lipasemedicine.diseaseLipoprotein LipaseEndocrinologychemistryPancreatitisMutationPancreatitisBiomarkersAtherosclerosis
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Transient chylomicronemia preceding the onset of insulin-dependent diabetes in a young girl with no humoral markers of islet autoimmunity

2004

OBJECTIVE: We investigated the possible causes of diabetes in a young child who presented with hyperglycemia associated with severe hypertriglyceridemia (>166 mmol/l), hypercholesterolemia (>38 mmol/l) and fasting chilomicrons. RESULTS: The patient did not have any of the HLA and autoantibody markers typically associated with type 1 diabetes. A glucose clamp failed to demonstrate insulin resistance (peripheral glucose utilization rate (M)=4.3 mg/kg per min) and there was no family history of type 2 diabetes or maturity onset diabetes in youth. Both fasting and stimulated C-peptide levels, including those in response to i.v. glucagon, were below the limit of detection. This is consiste…

medicine.medical_specialtyEndocrinology Diabetes and MetabolismHypercholesterolemiaAutoimmunityType 2 diabeteschylomicronemia diabetes young girl autoimmunityGlucagonIslets of LangerhansLipoprotein lipase deficiencyEndocrinologyInsulin resistanceInternal medicineDiabetes mellitusChylomicronsmedicineHumansChildAutoantibodiesHypertriglyceridemiaType 1 diabetesC-Peptidebusiness.industryHypertriglyceridemiaFastingGeneral MedicineGlucose clamp techniqueGlucagonmedicine.diseaseLipoprotein LipaseDiabetes Mellitus Type 1EndocrinologyHyperglycemiaGlucose Clamp TechniqueFemalelipids (amino acids peptides and proteins)businessEuropean Journal of Endocrinology
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