NCT02255552

Brief Summary

The main objective of this study is to provide evidence of efficacy of eteplirsen (AVI-4658) in Duchenne muscular dystrophy (DMD) patients that are amenable to skipping exon 51. Additional objectives include evaluation of safety, biomarkers and the long-term effects of eteplirsen up to 96 weeks, followed by a safety extension (not to exceed 48 weeks).

Trial Health

87
On Track

Trial Health Score

Automated assessment based on enrollment pace, timeline, and geographic reach

Enrollment
109

participants targeted

Target at P25-P50 for phase_3

Timeline
Completed

Started Nov 2014

Longer than P75 for phase_3

Geographic Reach
1 country

37 active sites

Status
completed

Health score is calculated from publicly available data and should be used for screening purposes only.

Trial Relationships

Click on a node to explore related trials.

Study Timeline

Key milestones and dates

First Submitted

Initial submission to the registry

September 25, 2014

Completed
7 days until next milestone

First Posted

Study publicly available on registry

October 2, 2014

Completed
2 months until next milestone

Study Start

First participant enrolled

November 17, 2014

Completed
4.6 years until next milestone

Primary Completion

Last participant's last visit for primary outcome

June 14, 2019

Completed
Same day until next milestone

Study Completion

Last participant's last visit for all outcomes

June 14, 2019

Completed
1 year until next milestone

Results Posted

Study results publicly available

July 1, 2020

Completed
Last Updated

January 25, 2021

Status Verified

December 1, 2020

Enrollment Period

4.6 years

First QC Date

September 25, 2014

Results QC Date

June 12, 2020

Last Update Submit

December 31, 2020

Conditions

Keywords

DMD, Duchenne, Eteplirsen, dystrophy, dystrophin, exon 51

Outcome Measures

Primary Outcomes (1)

  • Change From Baseline in the 6 Minute Walk Test (6MWT) Distance at Week 96

    6MWT was performed by standardized procedures for all participants. Participants were asked to walk a set course of 25 meters for 6 minutes (timed), and the distance walked (in meters) was recorded. Change from baseline in 6MWT distance at Week 96 was reported.

    Baseline, Week 96

Secondary Outcomes (6)

  • Change From Baseline in Dystrophin Protein Levels Determined by Western Blot at Week 96

    Baseline, Week 96

  • Number of Participants Having Ability to Rise Independently From the Floor Determined Based on North Star Ambulatory Assessment (NSAA) at Week 96

    Week 96

  • Number of Participants Who Lost Ambulation (LOA) by Week 96

    Up to Week 96

  • Change From Baseline in Forced Vital Capacity Percent (FVC%) Predicted at Weeks 96

    Baseline, Week 96

  • Change From Baseline in North Star Ambulatory Assessment (NSAA) Total Scores at Week 96

    Baseline, Week 96

  • +1 more secondary outcomes

Study Arms (2)

Treated Group

EXPERIMENTAL

Approximately 80 patients with genotypically confirmed Duchenne muscular dystrophy (DMD) with genetic deletions amenable to treatment by exon 51 skipping will receive 30 mg/kg of eteplirsen weekly for 96 weeks, followed by a safety extension (not to exceed 48 weeks).

Drug: eteplirsen

Untreated Group

NO INTERVENTION

Approximately 30 DMD patients not amenable to exon 51 skipping will not receive eteplirsen.

Interventions

Eteplirsen 30 mg/kg will be administered as an IV infusion once a week for 96 weeks, followed by a safety extension (not to exceed 48 weeks).

Also known as: AVI-4658, EXONDYS 51®
Treated Group

Eligibility Criteria

Age7 Years - 16 Years
Sexmale
Healthy VolunteersNo
Age GroupsChild (0-17)

You may qualify if:

  • Male 7-16 years old
  • Diagnosed with DMD, genotypically confirmed
  • Stable dose of corticosteroids for at least 24 weeks
  • Have intact right and left alternative upper muscle groups
  • Mean 6MWT greater than 300m (primary analysis on 300 to 450 meters)
  • Stable pulmonary and cardiac function: predicted FVC equal to or greater than 50% and LVEF of greater than 50%

You may not qualify if:

  • Previous treatment with drisapersen or any other RNA antisense agent or any gene therapy within the last 6 months
  • Participation in any other DMD interventional clinical study within 12 weeks
  • Major surgery within 3 months
  • Presence of other clinically significant illness
  • Major change in the physical therapy regime within 3 months

Contact the study team to confirm eligibility.

Sponsors & Collaborators

Study Sites (37)

Neuromuscular Research Center

Phoenix, Arizona, 85028, United States

Location

David Geffen School of Medicine at UCLA

Los Angeles, California, 90095, United States

Location

University of California, Davis Medical Center

Sacramento, California, 95817, United States

Location

Rady Children's Hospital, U.C. San Diego

San Diego, California, 92130, United States

Location

Stanford University School of Medicine/Medical Center

Stanford, California, 94305, United States

Location

Children's Hospital Colorado

Aurora, Colorado, 80045, United States

Location

Connecticut Children's Medical Center

Hartford, Connecticut, 06106, United States

Location

Children's National Health System

Washington D.C., District of Columbia, 20010, United States

Location

The University of Florida, Powell Gene Therapy Center

Gainesville, Florida, 32610, United States

Location

NW FL Clinical Research Group, LLC

Gulf Breeze, Florida, 32561, United States

Location

Nemours Children's Hospital

Orlando, Florida, 32827, United States

Location

Rare Disease Research Center

Atlanta, Georgia, 30318, United States

Location

Emory University

Atlanta, Georgia, 30324, United States

Location

Ann and Robert H. Lurie Children's Hospital of Chicago

Chicago, Illinois, 60611, United States

Location

University of Iowa Children's Hospital

Iowa City, Iowa, 52242, United States

Location

University of Kansas Medical Center

Kansas City, Kansas, 66160, United States

Location

Kennedy Krieger Institute

Baltimore, Maryland, 21205, United States

Location

Massachusetts General Hospital

Boston, Massachusetts, 02114, United States

Location

Children's Hospital Boston

Boston, Massachusetts, 02115, United States

Location

Children's Hospital of Michigan

Detroit, Michigan, 48201, United States

Location

University of Minnesota

Minneapolis, Minnesota, 55455, United States

Location

St. Louis Children's Hospital

St Louis, Missouri, 63110, United States

Location

Dartmouth-Hitchcock Medical Center

Lebanon, New Hampshire, 03756, United States

Location

Columbia University Medical Center

New York, New York, 10032, United States

Location

University of Rochester Clinical Research Center

Rochester, New York, 14642, United States

Location

Levine Childrens Hospital, Carolinas Medical Center

Charlotte, North Carolina, 28207, United States

Location

Cincinnati Children's Hospital Medical Center (CCHMC)

Cincinnati, Ohio, 45229, United States

Location

Nationwide Children's Hospital

Columbus, Ohio, 43205, United States

Location

Shriners Hospital for Children

Portland, Oregon, 97239, United States

Location

Penn State Hershey Medical Center

Hershey, Pennsylvania, 17033, United States

Location

Children's Hospital of Philadelphia

Philadelphia, Pennsylvania, 19104, United States

Location

Children's Hospital of Pittsburgh of UPMC

Pittsburgh, Pennsylvania, 15224, United States

Location

Vanderbilt University Medical Center

Nashville, Tennessee, 37232, United States

Location

The University of Texas Southwestern Medical Center

Dallas, Texas, 75390, United States

Location

Texas Children's Hospital

Houston, Texas, 77030, United States

Location

University of Utah

Salt Lake City, Utah, 84132, United States

Location

Seattle Children's Hospital

Seattle, Washington, 98105, United States

Location

Related Publications (1)

  • Brogna C, Coratti G, Pane M, Ricotti V, Messina S, D'Amico A, Bruno C, Vita G, Berardinelli A, Mazzone E, Magri F, Ricci F, Mongini T, Battini R, Bello L, Pegoraro E, Baranello G, Previtali SC, Politano L, Comi GP, Sansone VA, Donati A, Bertini E, Muntoni F, Goemans N, Mercuri E; on behalf on the International DMD group. Long-term natural history data in Duchenne muscular dystrophy ambulant patients with mutations amenable to skip exons 44, 45, 51 and 53. PLoS One. 2019 Jun 25;14(6):e0218683. doi: 10.1371/journal.pone.0218683. eCollection 2019.

    PMID: 31237898BACKGROUND

MeSH Terms

Conditions

Muscular Dystrophy, Duchenne

Interventions

eteplirsen

Condition Hierarchy (Ancestors)

Muscular DystrophiesMuscular Disorders, AtrophicMuscular DiseasesMusculoskeletal DiseasesNeuromuscular DiseasesNervous System DiseasesGenetic Diseases, X-LinkedGenetic Diseases, InbornCongenital, Hereditary, and Neonatal Diseases and Abnormalities

Results Point of Contact

Title
Medical Director
Organization
Sarepta Therapeutics, Inc.

Study Officials

  • Medical Director

    Sarepta Therapeutics, Inc.

    STUDY DIRECTOR

Publication Agreements

PI is Sponsor Employee
No
Restriction Type
OTHER
Restrictive Agreement
Yes

Study Design

Study Type
interventional
Phase
phase 3
Allocation
NON RANDOMIZED
Masking
NONE
Purpose
TREATMENT
Intervention Model
PARALLEL
Sponsor Type
INDUSTRY
Responsible Party
SPONSOR

Study Record Dates

First Submitted

September 25, 2014

First Posted

October 2, 2014

Study Start

November 17, 2014

Primary Completion

June 14, 2019

Study Completion

June 14, 2019

Last Updated

January 25, 2021

Results First Posted

July 1, 2020

Record last verified: 2020-12

Data Sharing

IPD Sharing
Will not share

Locations