Rationale and design of two trials assessing the efficacy, safety, and tolerability of inclisiran in adolescents with homozygous and heterozygous familial hypercholesterolaemia.


Journal

European journal of preventive cardiology
ISSN: 2047-4881
Titre abrégé: Eur J Prev Cardiol
Pays: England
ID NLM: 101564430

Informations de publication

Date de publication:
20 07 2022
Historique:
received: 17 11 2021
revised: 31 01 2022
pubmed: 18 2 2022
medline: 23 7 2022
entrez: 17 2 2022
Statut: ppublish

Résumé

Inclisiran is a small interfering RNA molecule that reduces low-density lipoprotein cholesterol (LDL-C) by inhibition of proprotein convertase subtilisin/kexin type 9. This subcutaneous, twice-yearly administered agent has been shown to effectively and safely lower LDL-C in adult patients with established atherosclerotic cardiovascular disease, adults at high risk for atherosclerotic cardiovascular disease, as well as in adults with heterozygous familial hypercholesterolaemia. With the current, limited treatment options available to reach treatment goals in children with severe heterozygous familial hypercholesterolaemia, homozygous familial hypercholesterolaemia, or statin intolerance, inclisiran could be a valuable new therapeutic option. The objective of these ongoing studies is to investigate the efficacy, safety, and tolerability of inclisiran in adolescents diagnosed with homozygous familial hypercholesterolaemia (ORION-13) or heterozygous familial hypercholesterolaemia (ORION-16). ORION-13 and ORION-16 are both two-part (1-year double-blind inclisiran vs. placebo/1 year open-label inclisiran) multicentre trials including adolescents aged 12 to <18 years diagnosed with familial hypercholesterolaemia. ORION-13 will include ∼12 participants diagnosed with homozygous familial hypercholesterolaemia and ORION-16 will include ∼150 participants diagnosed with heterozygous familial hypercholesteroleamia. The primary endpoint is the percentage change in LDL-C from baseline to Day 330. Secondary efficacy and safety endpoints include changes in other lipid parameters and treatment-emergent adverse events as well as laboratory parameters and vital signs. Exploratory endpoints include individual responsiveness of the participants and change in LDL-C according to the type of underlying causal mutation. https://www.clinicaltrials.gov/. Unique identifier: NCT04659863 (ORION-13) and NCT04652726 (ORION-16).

Sections du résumé

BACKGROUND
Inclisiran is a small interfering RNA molecule that reduces low-density lipoprotein cholesterol (LDL-C) by inhibition of proprotein convertase subtilisin/kexin type 9. This subcutaneous, twice-yearly administered agent has been shown to effectively and safely lower LDL-C in adult patients with established atherosclerotic cardiovascular disease, adults at high risk for atherosclerotic cardiovascular disease, as well as in adults with heterozygous familial hypercholesterolaemia. With the current, limited treatment options available to reach treatment goals in children with severe heterozygous familial hypercholesterolaemia, homozygous familial hypercholesterolaemia, or statin intolerance, inclisiran could be a valuable new therapeutic option.
OBJECTIVES
The objective of these ongoing studies is to investigate the efficacy, safety, and tolerability of inclisiran in adolescents diagnosed with homozygous familial hypercholesterolaemia (ORION-13) or heterozygous familial hypercholesterolaemia (ORION-16).
STUDY DESIGN
ORION-13 and ORION-16 are both two-part (1-year double-blind inclisiran vs. placebo/1 year open-label inclisiran) multicentre trials including adolescents aged 12 to <18 years diagnosed with familial hypercholesterolaemia. ORION-13 will include ∼12 participants diagnosed with homozygous familial hypercholesterolaemia and ORION-16 will include ∼150 participants diagnosed with heterozygous familial hypercholesteroleamia. The primary endpoint is the percentage change in LDL-C from baseline to Day 330. Secondary efficacy and safety endpoints include changes in other lipid parameters and treatment-emergent adverse events as well as laboratory parameters and vital signs. Exploratory endpoints include individual responsiveness of the participants and change in LDL-C according to the type of underlying causal mutation.
CLINICAL TRIAL REGISTRATION
https://www.clinicaltrials.gov/. Unique identifier: NCT04659863 (ORION-13) and NCT04652726 (ORION-16).

Identifiants

pubmed: 35175352
pii: 6530162
doi: 10.1093/eurjpc/zwac025
doi:

Substances chimiques

ALN-PCS 0
Anticholesteremic Agents 0
Cholesterol, LDL 0
RNA, Small Interfering 0

Banques de données

ClinicalTrials.gov
['NCT04659863', 'NCT04652726']

Types de publication

Journal Article Randomized Controlled Trial Research Support, Non-U.S. Gov't

Langues

eng

Sous-ensembles de citation

IM

Pagination

1361-1368

Informations de copyright

Published on behalf of the European Society of Cardiology. All rights reserved. © The Author(s) 2022. For permissions, please email: journals.permissions@oup.com.

Auteurs

M Doortje Reijman (MD)

Department of Paediatrics, Amsterdam UMC, Location AMC, Meibergdreef 9, 1105 AZ Amsterdam, The Netherlands.

Anja Schweizer (A)

Global Drug Development, Cardiovascular, Renal and Metabolism, Novartis Pharma AG, Basel, Switzerland.

Amy L H Peterson (ALH)

Division of Paediatric Cardiology, Department of Paediatrics, University of Wisconsin School of Medicine and Public Health, Madison, WI, USA.

Eric Bruckert (E)

Department of Endocrinology and Prevention of Cardiovascular Disease, Pitié-Salpêtrière University Hospital, Paris, France.

Christian Stratz (C)

Global Drug Development, Cardiovascular, Renal and Metabolism, Novartis Pharma AG, Basel, Switzerland.

Joep C Defesche (JC)

Department of Clinical Genetics, Amsterdam UMC, Location AMC, University of Amsterdam, Amsterdam, The Netherlands.

Robert A Hegele (RA)

Department of Medicine, Schulich School of Medicine and Dentistry, Western University, London, Ontario, Canada.

Albert Wiegman (A)

Department of Paediatrics, Amsterdam UMC, Location AMC, Meibergdreef 9, 1105 AZ Amsterdam, The Netherlands.

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Classifications MeSH