Long-term safety and efficacy of lomitapide in patients with homozygous familial hypercholesterolemia: Five-year data from the Lomitapide Observational Worldwide Evaluation Registry (LOWER).


Journal

Journal of clinical lipidology
ISSN: 1933-2874
Titre abrégé: J Clin Lipidol
Pays: United States
ID NLM: 101300157

Informations de publication

Date de publication:
Historique:
received: 03 02 2020
revised: 06 08 2020
accepted: 12 08 2020
pubmed: 8 10 2020
medline: 27 8 2021
entrez: 7 10 2020
Statut: ppublish

Résumé

Lomitapide is a lipid-lowering agent indicated as adjunct therapy for homozygous familial hypercholesterolemia (HoFH) in adults. The Lomitapide Observational Worldwide Evaluation Registry is an international, observational registry assessing long-term safety, tolerability, and effectiveness of lomitapide. This analysis examines 5-year data from the registry up to February 28, 2019. At lomitapide initiation, enrolled patients (N = 187) were a mean ± SD age of 52.2 ± 15.3 years with a mean ± SD low-density lipoprotein cholesterol (LDL-C) measurement of 232.0 ± 94.9 mg/dL. Exposure duration was up to 5.9 years (median, 1.98 years), and median dose was 10 mg (range, 5 mg QOD to 40 mg QD). After treatment, there was a mean 33% reduction in LDL-C (45% in patients remaining on lomitapide), 65.4% achieved LDL-C <100 mg/dL, and 41.1% achieved LDL-C <70 mg/dL. At year 4, the absolute mean change from baseline in LDL-C was -70.6 ± 76.21 mg/dL. Adverse events (AEs) occurred in 75.7% of patients, treatment-related AEs in 54.6%, and serious AEs in 22.2%; 23.2% of patients discontinued because of an AE. Events of special interest included gastrointestinal (13.5%), hepatic (15.1%), major adverse cardiovascular events (10.8%, resulting in 5 deaths), tumors (2.2%), and 4 pregnancies in 3 of 32 women of childbearing potential. The efficacy and safety of lomitapide are consistent with phase III trial data despite using a much lower median dose of 10 mg vs 40 mg in phase III. No new safety signals were identified. The incidence of AEs, serious AEs, and aminotransferase alanine transaminase elevations was lower than that seen in the phase III trial, potentially related to the lower median dose.

Sections du résumé

BACKGROUND
Lomitapide is a lipid-lowering agent indicated as adjunct therapy for homozygous familial hypercholesterolemia (HoFH) in adults.
OBJECTIVE
The Lomitapide Observational Worldwide Evaluation Registry is an international, observational registry assessing long-term safety, tolerability, and effectiveness of lomitapide.
METHODS
This analysis examines 5-year data from the registry up to February 28, 2019.
RESULTS
At lomitapide initiation, enrolled patients (N = 187) were a mean ± SD age of 52.2 ± 15.3 years with a mean ± SD low-density lipoprotein cholesterol (LDL-C) measurement of 232.0 ± 94.9 mg/dL. Exposure duration was up to 5.9 years (median, 1.98 years), and median dose was 10 mg (range, 5 mg QOD to 40 mg QD). After treatment, there was a mean 33% reduction in LDL-C (45% in patients remaining on lomitapide), 65.4% achieved LDL-C <100 mg/dL, and 41.1% achieved LDL-C <70 mg/dL. At year 4, the absolute mean change from baseline in LDL-C was -70.6 ± 76.21 mg/dL. Adverse events (AEs) occurred in 75.7% of patients, treatment-related AEs in 54.6%, and serious AEs in 22.2%; 23.2% of patients discontinued because of an AE. Events of special interest included gastrointestinal (13.5%), hepatic (15.1%), major adverse cardiovascular events (10.8%, resulting in 5 deaths), tumors (2.2%), and 4 pregnancies in 3 of 32 women of childbearing potential.
CONCLUSION
The efficacy and safety of lomitapide are consistent with phase III trial data despite using a much lower median dose of 10 mg vs 40 mg in phase III. No new safety signals were identified. The incidence of AEs, serious AEs, and aminotransferase alanine transaminase elevations was lower than that seen in the phase III trial, potentially related to the lower median dose.

Identifiants

pubmed: 33023859
pii: S1933-2874(20)30251-8
doi: 10.1016/j.jacl.2020.08.006
pii:
doi:

Substances chimiques

BMS201038 0
Benzimidazoles 0

Types de publication

Journal Article Observational Study Research Support, Non-U.S. Gov't

Langues

eng

Sous-ensembles de citation

IM

Pagination

807-817

Informations de copyright

Copyright © 2020 National Lipid Association. Published by Elsevier Inc. All rights reserved.

Auteurs

James A Underberg (JA)

Department of Medicine, New York University Langone Medical Center, New York, NY, USA. Electronic address: James.Underberg@nyulangone.org.

Christopher P Cannon (CP)

Department of Medicine, Baim Institute for Clinical Research, Boston, MA, USA.

Dominique Larrey (D)

Liver and Transplantation Unit, Montpellier School of Medicine and IRB-INSERM 11183, Montpellier, France.

Lukas Makris (L)

Stathmi, Inc, New Hope, PA, USA.

Dirk Blom (D)

Division of Lipidology and Hatter Institute for Cardiovascular Research in Southern Africa, Department of Medicine, University of Cape Town, Cape Town, South Africa.

Helen Phillips (H)

Amryt Pharma plc, London, England, United Kingdom.

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