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FUSION: A Study to Evaluate the Efficacy, Safety, Pharmacokinetics and Pharmacodynamics of ION363 in Amyotrophic Lateral Sclerosis Participants With Fused in Sarcoma Mutations (FUS-ALS)

Phase 3
Recruiting
Conditions
Amyotrophic Lateral Sclerosis
Interventions
Drug: Placebo
Drug: ION363
Registration Number
NCT04768972
Lead Sponsor
Ionis Pharmaceuticals, Inc.
Brief Summary

The primary purpose of this study is to evaluate the efficacy of ION363 on clinical function and survival in carriers of fused in sarcoma mutations with amyotrophic lateral sclerosis (FUS-ALS).

Detailed Description

This is a multi-center, three-part study of ION363 in up to 95 participants. Part 1 will consist of participants who will be randomized in a 2:1 ratio to receive a multi-dose regimen of ION363 or placebo for a period of 60 weeks, followed by Part 2, in which participants will receive open-label ION363 for a period of 84 weeks. Participants may continue to receive open-label ION363 in Part 3 for up to 3 additional years or until ION363 becomes commercially available in the patient's country or until the Sponsor discontinues the ION363 development program, whichever occurs earlier.

Recruitment & Eligibility

Status
RECRUITING
Sex
All
Target Recruitment
95
Inclusion Criteria

Not provided

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Exclusion Criteria

Not provided

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Study & Design

Study Type
INTERVENTIONAL
Study Design
PARALLEL
Arm && Interventions
GroupInterventionDescription
PlaceboPlaceboPlacebo will be administered by lumbar IT bolus injection every12 weeks, with an additional loading dose at 4 weeks, over a 60-week double-blind treatment period (Part 1).
ION363ION363ION363 will be administered by lumbar intrathecal (IT) bolus injection every 12 weeks, with an additional loading dose at 4 weeks, over a 60-week double-blind treatment period in Part 1; every 12 weeks for 84 weeks in the open-label extension treatment period (Part 2), with an additional loading dose administered 4 weeks after the first dose. Patients may continue to receive open-label ION363 every 12 weeks in Part 3 for up to 3 additional years or until ION363 becomes commercially available in the patient's country or until the Sponsor discontinues the development program, whichever occurs earlier.
Primary Outcome Measures
NameTimeMethod
Change from Baseline (Day 1) through Study Day 505 in Part 1 in functional impairmentBaseline, Day 505 in Part 1

Functional impairment to be measured by joint rank analysis of the combined assessment of: In-clinic Amyotrophic Lateral Sclerosis Functional Rating Scale-Revised (ALSFRS-R) total score, time of rescue or discontinuation from Part 1 and entering Part 2 due to a deterioration in function, and ventilation assistance-free survival (VAFS). ALSFRS-R measures functional disease severity. The scale measures four functional domains, bulbar function, gross motor skills, fine motor skills, and respiratory function. The assessment will contain 12 questions scored from 0 (no function) to 4 (full function), with a total possible score of 48, which will indicate the highest level of function. ALSFRS-R will be a part of the combined assessment of joint rank analysis to assess efficacy in Part 1.

Secondary Outcome Measures
NameTimeMethod
Change from Baseline in the in-clinic Amyotrophic Lateral Sclerosis Functional Rating Scale-Revised (ALSFRS-R)Baseline, Day 505 in Part 1
Survival and Ventilation Assistance-Free Survival (VAFS)Up to Day 505 in Part 1
Change from Baseline in Handheld Dynamometry (HHD) to Day 505 in Part 1Baseline, Day 505 in Part 1
Change from Baseline in Neurofilament Light (NfL) Concentration in Cerebrospinal Fluid (CSF) to Day 505Baseline, Day 505 in Part 1
Change from Baseline in In-clinic Slow Vital Capacity (SVC) to Day 505 in Part 1Baseline, Day 505 in Part 1
Change from Baseline in FUS Concentration in Cerebrospinal Fluid (CSF) to Day 505Baseline, Day 505 in Part 1
Change from Baseline in Amyotrophic Lateral Sclerosis Specific Quality of Life - Revised (ALSSQOL-R) Score to Day 505 in Part 1Baseline, Day 505 in Part 1

The ALSSQOL-R is a disease-specific 50-item assessment that measures the quality of life. Each item will be rated by the participant on a scale of 0 to 10, with 0 being the least desirable situation and 10 being the most desirable.

Trial Locations

Locations (25)

Seoul National University Hospital

馃嚢馃嚪

Seoul, Korea, Republic of

University of California San Diego

馃嚭馃嚫

La Jolla, California, United States

Stanford University Medical Center

馃嚭馃嚫

Palo Alto, California, United States

Johns Hopkins University

馃嚭馃嚫

Baltimore, Maryland, United States

Massachusetts General Hospital

馃嚭馃嚫

Boston, Massachusetts, United States

Washington University School of Medicine

馃嚭馃嚫

Saint Louis, Missouri, United States

Columbia University Medical Center

馃嚭馃嚫

New York, New York, United States

The Ohio State University Wexner Medical Center

馃嚭馃嚫

Columbus, Ohio, United States

University of Utah

馃嚭馃嚫

Salt Lake City, Utah, United States

UZ Leuven

馃嚙馃嚜

Leuven, VL-Brabant, Belgium

Hospital Universitari de Bellvitge

馃嚜馃嚫

Barcelona, Spain

University Hospital of Umea

馃嚫馃嚜

Ume氓, Sweden

Kantonsspital St. Gallen

馃嚚馃嚟

Saint Gallen, Switzerland

PSEG Centro de Pesquisa Clinica S.A.

馃嚙馃嚪

S茫o Paulo, Brazil

Montreal Neurological Institute

馃嚚馃嚘

Montreal, Quebec, Canada

Universitaetsmedizin Rostock

馃嚛馃嚜

Rostock, Germany

Universit盲tsklinikum Ulm

馃嚛馃嚜

Ulm, Germany

St. James Hospital

馃嚠馃嚜

Dublin, Ireland

Citta della Salute e della Scienza di Torino - Ospedale le Molinette

馃嚠馃嚬

Torino, Italy

Toho University Omori Medical Center

馃嚡馃嚨

Tokyo, Japan

Hanyang University Seoul Hospital

馃嚢馃嚪

Seoul, Korea, Republic of

Universitair Medisch Centrum Utrecht

馃嚦馃嚤

Utrecht, Netherlands

Linden sp贸lka z ograniczona odpowiedzialnoscia sp贸lka komandytow

馃嚨馃嚤

Krak贸w, Poland

Taipei Veterans General Hospital (VGHTP)

馃嚚馃嚦

Taipei City, Taiwan

King's College Hospital

馃嚞馃嚙

London, United Kingdom

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