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AVI-4658

Phase 2

Duchenne Muscular Dystrophy | Small molecule | Neurology |Sarepta Therapeutics, Inc.|Last Updated: Mar 30, 2020

Success Probability

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Market & Valuation

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Trial Design

RandomizedDouble-BlindCONTROLLEDDMC
Total Trials2
Total Enrollment31

FDA Designations

No designations recorded

Clinical trial landscape

AVI-4658 · 3 trials · 2 indications

Phase 2 2Phase 1 1
NCT01540409Efficacy, Safety, and Tolerability Rollover Study of Eteplirsen in Subjects With Duchenne Muscular DystrophyDuchenne Muscular Dystrophy (DMD)
COMPLETED12 Analytics
NCT01396239Efficacy Study of AVI-4658 to Induce Dystrophin Expression in Selected Duchenne Muscular Dystrophy PatientsDuchenne Muscular Dystrophy
COMPLETED12 Analytics
PHASE2COMPLETED
Efficacy, Safety, and Tolerability Rollover Study of Eteplirsen in Subjects With Duchenne Muscular Dystrophy
Duchenne Muscular Dystrophy (DMD)Unlock trial analytics
PHASE2COMPLETED
Efficacy Study of AVI-4658 to Induce Dystrophin Expression in Selected Duchenne Muscular Dystrophy Patients
Duchenne Muscular DystrophyUnlock trial analytics

Study Endpoints

Primary Endpoints

Change From Baseline in the 6 Minute Walk Test (6MWT) at Week 240
Parent Baseline and Week 240

This study used a modified version of the 6MWT test procedure described in American Thoracic Society (ATS) 2002 guidelines, specifically adapted for patients with Duchenne muscular dystrophy. The participant was asked to walk a set course of 25 meters for 6 minutes (timed) and the distance walked in meters was recorded. Increases from baseline in 6MWT distance are indicative of improvement and decreases from baseline indicate worsening. Baseline here corresponds to the baseline in the parent study (4658-us-201, NCT01396239).

Change From Baseline in the Percentage of Dystrophin Positive Fibers (PDPF) at Week 48
Parent Baseline and Week 48

Dystrophin expression as assessed by percent dystrophin positive fibers was measured by immunohistochemistry (IHC) technique using primary anti-dystrophin antibody. Percent change from baseline is the arithmetic difference of the treatment time point minus baseline divided by baseline calculated for individual subjects. Baseline here corresponds to the baseline in the parent study (4658-us-201, NCT01396239).

Change in the Number (%) of Dystrophin Positive Fibers
After 12 weeks for 4 patients who received 50 mg/kg and 2 patients who received placebo. After 24 weeks for 4 patients who received 30 mg/kg and 2 patients who received placebo.

The primary efficacy endpoint will be based on the pre-treatment and post-treatment change in the number (%) of dystrophin positive fibers as measured in the muscle biopsy tissue on immunohistochemistry (IHC).

Safety and Tolerability
Baseline to 6 months

Number of subjects with 1 or more Treatment Emergent Adverse Event that are possibly related to the investigational drug

Treatment Emergent Adverse Events
from Baseline to Follow up (27 weeks)

Number of Patients with Treatment Emergent Adverse Events

Secondary Endpoints

Change From Baseline: 6 Minute Walk Test (6MWT) - Intent to Treat Population (ITT)
24 weeks
Change From Baseline: 6 Minute Walk Test (6MWT) - Modified Intent to Treat Population (mITT)
24 weeks
Pharmacokinetics - Mean Peak Plasma Concentration of AVI-4658 After Administration
Samples were taken: 30 minutes pre dose; and at 5 (±1), 15 (±2), 30 (±5), 60 (±5), and 90 (±5) minutes; and 2, 4, 6, 8, 12, and 24 hours (all ± 15 minutes) post dose at Weeks 1, 6, and 12
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Study Design & Arms

AllocationNA
MaskingNONE
ModelSINGLE_GROUP
PurposeTREATMENT

Treatment Arms

ArmTypeDescription
AVI-4658 (Eteplirsen)EXPERIMENTALMultiple-Dose Extension Study
Placebo / Delayed TreatmentPLACEBO_COMPARATOR3a. Placebo 50 mg/kg phosphate buffered saline solution identical in appearance to eteplirsen for 24 weeks followed by 50 mg/kg of eteplirsen for 4 weeks. 3b. Placebo 30 mg/kg phosphate buffered saline solution identical in appearance to eteplirsen for 24 weeks followed by 30 mg/kg of eteplirsen for 4 weeks.
Cohort 1 - 0.5 mg/kg/wkEXPERIMENTALSubjects in this group will receive a 0.5 mg/kg/wk dose of AVI-4658 over 12 weekly IV infusions in 50 mL of normal saline solution over a 60-minute period
Cohort 2 - 1.0 mg/kg/wkEXPERIMENTALSubjects in this group will receive a 1.0 mg/kg/wk dose of AVI-4658 over 12 weekly IV infusions in 50 mL of normal saline solution over a 60-minute period
Cohort 3 - 2.0 mg/kg/wkEXPERIMENTALSubjects in this group will receive a 2.0 mg/kg/wk dose of AVI-4658 over 12 weekly IV infusions in 50 mL of normal saline solution over a 60-minute period
Cohort 4 - 4.0 mg/kg/wkEXPERIMENTALSubjects in this group will receive a 4.0 mg/kg/wk dose of AVI-4658 over 12 weekly IV infusions in 50 mL of normal saline solution over a 60-minute period
Cohort 5 - 10.0 mg/kg/wkEXPERIMENTALSubjects in this group will receive a 10.0 mg/kg/wk dose of AVI-4658 over 12 weekly IV infusions in 50 mL of normal saline solution over a 60-minute period
Cohort 6 - 20.0 mg/kg/wkEXPERIMENTALSubjects in this group will receive a 20.0 mg/kg/wk dose of AVI-4658 over 12 weekly IV infusions in 50 mL of normal saline solution over a 60-minute period

Interventions

NameTypeDescription
AVI-4658 (Eteplirsen)DRUGEteplirsen will be administered once weekly via an IV infusion. There are two treatment groups, 30 mg/kg and 50 mg/kg.
PlaceboOTHERsterile, isotonic, clear, colorless phosphate buffered saline solution of eteplirsen at a concentration of 100 mg/mL in single-use vials containing a nominal volume of 1.0 mL without preservatives.
AVI-4658 for InjectionDRUGAVI-4658 for Injection, is packaged as 100 mg/mL in phosphate buffered saline with 1 mL per vial. Study dosages will be infused over a 1 hour period with Normal saline in 6 dose cohorts.
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Eligibility Criteria

Age Range7 Years to 13 Years
SexMALE
Healthy VolunteersNo
Study Sites11

Inclusion Criteria: A subject must meet all of the following criteria to be eligible for this study. 1. The subject and/or their parent/legal guardian are willing and able to provide signed informed consent. 2. The subject has successfully completed 28 weeks of treatment in Study 4658-US-201. 3. T...

Countries:United StatesUnited Kingdom
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Frequently asked questions about AVI-4658

What is AVI-4658 used for in Duchenne Muscular Dystrophy?

AVI-4658 is an investigational small molecule being developed for the treatment of Duchenne Muscular Dystrophy (DMD), a genetic disorder characterized by progressive muscle degeneration. It is designed to induce dystrophin expression in selected DMD patients. The drug is currently in clinical development and has not been approved by regulatory authorities.

Who makes AVI-4658?

AVI-4658 is being developed by Sarepta Therapeutics, Inc., a biopharmaceutical company traded on the NASDAQ under the ticker symbol SRPT. Sarepta focuses on developing RNA-targeted therapeutics for rare neuromuscular diseases, including Duchenne Muscular Dystrophy.

What phase is AVI-4658 in?

AVI-4658 is in Phase 2 clinical development for Duchenne Muscular Dystrophy. It has completed two clinical trials, including a Phase 1 dose-ranging study and a Phase 2 efficacy study. The drug remains investigational and has not received FDA approval.

What clinical trials is AVI-4658 in?

AVI-4658 has completed two clinical trials: NCT00844597, a Phase 1 dose-ranging study in the United Kingdom with 19 participants, and NCT01396239, a Phase 2 efficacy study in the United States with 12 participants. Both trials enrolled male patients with Duchenne Muscular Dystrophy.

Is AVI-4658 the same as eteplirsen?

AVI-4658 is also known as eteplirsen, as indicated by the clinical trial NCT01540409, which is a rollover study of eteplirsen in subjects with Duchenne Muscular Dystrophy. This trial enrolled 12 participants and was completed in the United States.