Trial Outcomes & Findings for Safety Study of Eteplirsen to Treat Advanced Stage Duchenne Muscular Dystrophy (NCT NCT02286947)

NCT ID: NCT02286947

Last Updated: 2020-03-30

Results Overview

An adverse event (AE) was any untoward medical occurrence in a participant that did not necessarily have a causal relationship with the study drug. A serious adverse event (SAE) was an AE resulting in any of the following outcomes: death; Life-threatening event; Required or prolonged inpatient hospitalization; persistent or significant disability/incapacity; congenital anomaly. Treatment-emergent AEs were events that developed or worsened during the on-treatment period (defined as time from first dose of study drug and up to 28 days after last dose of study drug (up to 100 weeks) that were absent before treatment or that worsened relative to pretreatment state. AEs included both serious and non-serious adverse events.

Recruitment status

COMPLETED

Study phase

PHASE2

Target enrollment

24 participants

Primary outcome timeframe

From first dose of drug up to 100 weeks

Results posted on

2020-03-30

Participant Flow

The study was conducted at 9 sites in the United States from November 2014 to March 2018.

Participant milestones

Participant milestones
Measure
Eteplirsen 30 mg/kg
Participants received eteplirsen 30 mg/kg intravenous (IV) infusions, once weekly, for 96 weeks.
Overall Study
STARTED
24
Overall Study
COMPLETED
22
Overall Study
NOT COMPLETED
2

Reasons for withdrawal

Reasons for withdrawal
Measure
Eteplirsen 30 mg/kg
Participants received eteplirsen 30 mg/kg intravenous (IV) infusions, once weekly, for 96 weeks.
Overall Study
Withdrawal by Subject
2

Baseline Characteristics

Safety Study of Eteplirsen to Treat Advanced Stage Duchenne Muscular Dystrophy

Baseline characteristics by cohort

Baseline characteristics by cohort
Measure
Eteplirsen 30 mg/kg
n=24 Participants
Participants received eteplirsen 30 mg/kg IV infusions, weekly, for 96 weeks.
Age, Continuous
12.9 years
STANDARD_DEVIATION 3.30 • n=99 Participants
Sex: Female, Male
Female
0 Participants
n=99 Participants
Sex: Female, Male
Male
24 Participants
n=99 Participants
Ethnicity (NIH/OMB)
Hispanic or Latino
3 Participants
n=99 Participants
Ethnicity (NIH/OMB)
Not Hispanic or Latino
20 Participants
n=99 Participants
Ethnicity (NIH/OMB)
Unknown or Not Reported
1 Participants
n=99 Participants
Race (NIH/OMB)
American Indian or Alaska Native
0 Participants
n=99 Participants
Race (NIH/OMB)
Asian
1 Participants
n=99 Participants
Race (NIH/OMB)
Native Hawaiian or Other Pacific Islander
0 Participants
n=99 Participants
Race (NIH/OMB)
Black or African American
0 Participants
n=99 Participants
Race (NIH/OMB)
White
21 Participants
n=99 Participants
Race (NIH/OMB)
More than one race
2 Participants
n=99 Participants
Race (NIH/OMB)
Unknown or Not Reported
0 Participants
n=99 Participants
Region of Enrollment
United States
24 participants
n=99 Participants

PRIMARY outcome

Timeframe: From first dose of drug up to 100 weeks

Population: Analysis was performed on safety population included all participants who received at least 1 dose of eteplirsen.

An adverse event (AE) was any untoward medical occurrence in a participant that did not necessarily have a causal relationship with the study drug. A serious adverse event (SAE) was an AE resulting in any of the following outcomes: death; Life-threatening event; Required or prolonged inpatient hospitalization; persistent or significant disability/incapacity; congenital anomaly. Treatment-emergent AEs were events that developed or worsened during the on-treatment period (defined as time from first dose of study drug and up to 28 days after last dose of study drug (up to 100 weeks) that were absent before treatment or that worsened relative to pretreatment state. AEs included both serious and non-serious adverse events.

Outcome measures

Outcome measures
Measure
Eteplirsen 30 mg/kg
n=24 Participants
Participants received eteplirsen 30 mg/kg IV infusions, weekly, for 96 weeks.
Number of Participants With Treatment Emergent Adverse Events
24 Participants

SECONDARY outcome

Timeframe: Baseline up to 100 weeks

Population: Analysis was performed on safety population included all participants who received at least 1 dose of eteplirsen.

Laboratory parameters included hematology, clinical chemistry, urinalysis and coagulation. Data is only reported for parameters in which at least 1 participant had potentially clinically significant abnormal findings. Incr=increase; LLN=lower limit of normal; ULN=upper limit of normal; GGT=gamma glutamyl transferase

Outcome measures

Outcome measures
Measure
Eteplirsen 30 mg/kg
n=24 Participants
Participants received eteplirsen 30 mg/kg IV infusions, weekly, for 96 weeks.
Number of Participants With Potentially Clinically Significant Laboratory Abnormalities
Sodium: Decrease of 8 or more
3 Participants
Number of Participants With Potentially Clinically Significant Laboratory Abnormalities
Sodium: Increase of 8 or more
3 Participants
Number of Participants With Potentially Clinically Significant Laboratory Abnormalities
Potassium: Decrease of 1.1 or more
2 Participants
Number of Participants With Potentially Clinically Significant Laboratory Abnormalities
Potassium: Increase of 1.0 or more
2 Participants
Number of Participants With Potentially Clinically Significant Laboratory Abnormalities
Potassium: Value > 5.5 or < 3.0
1 Participants
Number of Participants With Potentially Clinically Significant Laboratory Abnormalities
Calcium: Decrease of 0.30 or more
1 Participants
Number of Participants With Potentially Clinically Significant Laboratory Abnormalities
Glucose: Decrease of 3.1 or more
1 Participants
Number of Participants With Potentially Clinically Significant Laboratory Abnormalities
Glucose: Increase of 3.2 or more
6 Participants
Number of Participants With Potentially Clinically Significant Laboratory Abnormalities
Albumin: Value < LLN or > ULN
4 Participants
Number of Participants With Potentially Clinically Significant Laboratory Abnormalities
Bilirubin: Incr of 10 or more
1 Participants
Number of Participants With Potentially Clinically Significant Laboratory Abnormalities
Bilirubin: Value > 1.5 x ULN
1 Participants
Number of Participants With Potentially Clinically Significant Laboratory Abnormalities
Alanine Aminotransferase: Value >= 2 x Baseline
1 Participants
Number of Participants With Potentially Clinically Significant Laboratory Abnormalities
GGT: value > 3*Baseline or > ULN
2 Participants
Number of Participants With Potentially Clinically Significant Laboratory Abnormalities
Lactate Dehydrogenase: Value >= 2 x Baseline
1 Participants
Number of Participants With Potentially Clinically Significant Laboratory Abnormalities
Creatine Kinase: Value >= 2 x Baseline
5 Participants
Number of Participants With Potentially Clinically Significant Laboratory Abnormalities
Hemoglobin: Value < LLN
5 Participants
Number of Participants With Potentially Clinically Significant Laboratory Abnormalities
Hematocrit: Value < LLN
7 Participants
Number of Participants With Potentially Clinically Significant Laboratory Abnormalities
Red Blood Cell: Value < LLN
4 Participants
Number of Participants With Potentially Clinically Significant Laboratory Abnormalities
White Blood Cell: Value < LLN or > 1.5 x ULN
5 Participants
Number of Participants With Potentially Clinically Significant Laboratory Abnormalities
Platelets: Value < 150 or < 200
1 Participants
Number of Participants With Potentially Clinically Significant Laboratory Abnormalities
Neutrophils: Value > 1.5 x ULN or < 1000
7 Participants
Number of Participants With Potentially Clinically Significant Laboratory Abnormalities
Lymphocytes: Value < LLN
6 Participants
Number of Participants With Potentially Clinically Significant Laboratory Abnormalities
Monocytes: Value < LLN
14 Participants
Number of Participants With Potentially Clinically Significant Laboratory Abnormalities
Eosinophils: Value > 1.5 x ULN or < LLN
2 Participants
Number of Participants With Potentially Clinically Significant Laboratory Abnormalities
Basophils: Value < LLN or > ULN
2 Participants
Number of Participants With Potentially Clinically Significant Laboratory Abnormalities
Urine Protein: Value > 1+
5 Participants

SECONDARY outcome

Timeframe: Baseline up to 100 weeks

Population: Analysis was performed on safety population included all participants who received at least 1 dose of eteplirsen.

Vital sign parameters included systolic blood pressure (SBP), diastolic blood pressure (DBP), heart rate (HR), and body temperature. Data is only reported for parameters in which at least 1 participant had potentially clinically significant abnormal vital sign findings.

Outcome measures

Outcome measures
Measure
Eteplirsen 30 mg/kg
n=24 Participants
Participants received eteplirsen 30 mg/kg IV infusions, weekly, for 96 weeks.
Number of Participants With Potentially Clinically Significant Abnormalities in Vital Signs
DBP: Less than 40 mmHG
1 Participants
Number of Participants With Potentially Clinically Significant Abnormalities in Vital Signs
DBP: Greater than 90 mmHg
12 Participants
Number of Participants With Potentially Clinically Significant Abnormalities in Vital Signs
SBP: Less than 80 mmHg
4 Participants
Number of Participants With Potentially Clinically Significant Abnormalities in Vital Signs
SBP: Greater than 130 mmHG
17 Participants
Number of Participants With Potentially Clinically Significant Abnormalities in Vital Signs
HR: Less than 50 beats per minute (bpm)
1 Participants
Number of Participants With Potentially Clinically Significant Abnormalities in Vital Signs
HR: Greater than 130 bpm
15 Participants

SECONDARY outcome

Timeframe: Baseline up to 100 weeks

Physical examinations, full and brief, were performed by the Investigator, a physician Sub-Investigator, or a Nurse Practitioner (if licensed in the state or province to perform physical examinations). Full physical examinations included examination of general appearance; head, ears, eyes, nose, and throat; heart; lungs; chest; abdomen; skin; lymph nodes; and musculoskeletal and neurological systems. Brief physical examinations included examination of general appearance; head, ears, eyes, nose, and throat; heart; lungs; chest; abdomen; and skin.

Outcome measures

Outcome measures
Measure
Eteplirsen 30 mg/kg
n=24 Participants
Participants received eteplirsen 30 mg/kg IV infusions, weekly, for 96 weeks.
Number of Participants With at Least One Potentially Clinically Significant Abnormalities in Physical Examinations
23 Participants

SECONDARY outcome

Timeframe: Baseline up to 100 weeks

Population: Analysis was performed on safety population included all participants who received at least 1 dose of eteplirsen.

Twelve-lead ECGs and Holter ECGs were performed at a consistent time of day throughout the study. Electrocardiograms were performed only after the patient was in the supine position, resting, and quiet for a minimum of 15 minutes. The ECG was manually reviewed and interpreted by medically qualified personnel using a central vendor according to prespecified criteria. The Investigator reviewed the results of the centrally read ECG report and determined if the findings were clinically significant. Data is only reported for parameters in which at least 1 participant had potentially clinically significant abnormal ECG findings. msec=milliseconds; QTcF=QT interval corrected with Fridericia's method

Outcome measures

Outcome measures
Measure
Eteplirsen 30 mg/kg
n=24 Participants
Participants received eteplirsen 30 mg/kg IV infusions, weekly, for 96 weeks.
Number of Participants With Abnormalities in Electrocardiograms (ECGs)
Heart Rate: >120 beats/minute
6 Participants
Number of Participants With Abnormalities in Electrocardiograms (ECGs)
QTcF: Increase of 60 or more msec
1 Participants

SECONDARY outcome

Timeframe: Baseline up to 100 weeks

Population: Analysis was performed on safety population included all participants who received at least 1 dose of eteplirsen.

Standard, 2-dimensional ECHOs were performed at a consistent time of day throughout the study. The ECHO was reviewed and interpreted by medically qualified personnel using a central vendor according to prespecified criteria. Ejection fraction was noted. The Investigator reviewed the results of the ECHO report and determined if the findings were clinically significant. LEVF=left ventricular ejection fraction

Outcome measures

Outcome measures
Measure
Eteplirsen 30 mg/kg
n=24 Participants
Participants received eteplirsen 30 mg/kg IV infusions, weekly, for 96 weeks.
Number of Participants With Abnormalities in Echocardiograms (ECHO)
LEVF: <55%
0 Participants
Number of Participants With Abnormalities in Echocardiograms (ECHO)
Fractional Shortening: <28%
6 Participants

Adverse Events

Eteplirsen 30 mg/kg

Serious events: 4 serious events
Other events: 24 other events
Deaths: 0 deaths

Serious adverse events

Serious adverse events
Measure
Eteplirsen 30 mg/kg
n=24 participants at risk
Participants received eteplirsen 30 mg/kg IV infusions, weekly, for 96 weeks.
Musculoskeletal and connective tissue disorders
Extremity contracture
4.2%
1/24 • Baseline up to Week 100
Musculoskeletal and connective tissue disorders
Scoliosis
4.2%
1/24 • Baseline up to Week 100
Cardiac disorders
Cardiomyopathy
4.2%
1/24 • Baseline up to Week 100
Renal and urinary disorders
Nephrolithiasis
4.2%
1/24 • Baseline up to Week 100

Other adverse events

Other adverse events
Measure
Eteplirsen 30 mg/kg
n=24 participants at risk
Participants received eteplirsen 30 mg/kg IV infusions, weekly, for 96 weeks.
Infections and infestations
Nasopharyngitis
58.3%
14/24 • Baseline up to Week 100
Infections and infestations
Upper respiratory tract infection
25.0%
6/24 • Baseline up to Week 100
Infections and infestations
Ear infection
12.5%
3/24 • Baseline up to Week 100
Infections and infestations
Gastroenteritis
12.5%
3/24 • Baseline up to Week 100
Infections and infestations
Pharyngitis streptococcal
12.5%
3/24 • Baseline up to Week 100
Respiratory, thoracic and mediastinal disorders
Cough
29.2%
7/24 • Baseline up to Week 100
Respiratory, thoracic and mediastinal disorders
Nasal congestion
20.8%
5/24 • Baseline up to Week 100
Respiratory, thoracic and mediastinal disorders
Oropharyngeal pain
20.8%
5/24 • Baseline up to Week 100
Respiratory, thoracic and mediastinal disorders
Rhinorrhoea
20.8%
5/24 • Baseline up to Week 100
Respiratory, thoracic and mediastinal disorders
Throat irritation
8.3%
2/24 • Baseline up to Week 100
Injury, poisoning and procedural complications
Contusion
25.0%
6/24 • Baseline up to Week 100
Injury, poisoning and procedural complications
Procedural pain
16.7%
4/24 • Baseline up to Week 100
Injury, poisoning and procedural complications
Joint injury
12.5%
3/24 • Baseline up to Week 100
Injury, poisoning and procedural complications
Ligament sprain
12.5%
3/24 • Baseline up to Week 100
Injury, poisoning and procedural complications
Foot fracture
8.3%
2/24 • Baseline up to Week 100
Injury, poisoning and procedural complications
Skin abrasion
8.3%
2/24 • Baseline up to Week 100
Injury, poisoning and procedural complications
Soft tissue injury
8.3%
2/24 • Baseline up to Week 100
Injury, poisoning and procedural complications
Spinal compression fracture
8.3%
2/24 • Baseline up to Week 100
Musculoskeletal and connective tissue disorders
Back pain
37.5%
9/24 • Baseline up to Week 100
Musculoskeletal and connective tissue disorders
Pain in extremity
16.7%
4/24 • Baseline up to Week 100
Musculoskeletal and connective tissue disorders
Arthralgia
12.5%
3/24 • Baseline up to Week 100
Musculoskeletal and connective tissue disorders
Muscle spasms
8.3%
2/24 • Baseline up to Week 100
Musculoskeletal and connective tissue disorders
Musculoskeletal pain
8.3%
2/24 • Baseline up to Week 100
Musculoskeletal and connective tissue disorders
Osteoporosis
8.3%
2/24 • Baseline up to Week 100
Gastrointestinal disorders
Vomiting
29.2%
7/24 • Baseline up to Week 100
Gastrointestinal disorders
Abdominal pain upper
25.0%
6/24 • Baseline up to Week 100
Gastrointestinal disorders
Diarrhoea
25.0%
6/24 • Baseline up to Week 100
Gastrointestinal disorders
Nausea
16.7%
4/24 • Baseline up to Week 100
Gastrointestinal disorders
Abdominal discomfort
12.5%
3/24 • Baseline up to Week 100
Gastrointestinal disorders
Constipation
12.5%
3/24 • Baseline up to Week 100
Skin and subcutaneous tissue disorders
Rash
33.3%
8/24 • Baseline up to Week 100
Skin and subcutaneous tissue disorders
Acne
8.3%
2/24 • Baseline up to Week 100
Skin and subcutaneous tissue disorders
Ecchymosis
8.3%
2/24 • Baseline up to Week 100
Skin and subcutaneous tissue disorders
Rash papular
8.3%
2/24 • Baseline up to Week 100
General disorders
Catheter site pain
20.8%
5/24 • Baseline up to Week 100
General disorders
Infusion site bruising
16.7%
4/24 • Baseline up to Week 100
General disorders
Catheter site bruise
12.5%
3/24 • Baseline up to Week 100
General disorders
Pyrexia
12.5%
3/24 • Baseline up to Week 100
General disorders
Chest pain
8.3%
2/24 • Baseline up to Week 100
General disorders
Infusion site pain
8.3%
2/24 • Baseline up to Week 100
Nervous system disorders
Headache
33.3%
8/24 • Baseline up to Week 100
Nervous system disorders
Dizziness
8.3%
2/24 • Baseline up to Week 100
Investigations
Breath sounds abnormal
12.5%
3/24 • Baseline up to Week 100
Investigations
Protein urine present
8.3%
2/24 • Baseline up to Week 100
Metabolism and nutrition disorders
Dehydration
8.3%
2/24 • Baseline up to Week 100
Psychiatric disorders
Depression
8.3%
2/24 • Baseline up to Week 100
Renal and urinary disorders
Renal pain
8.3%
2/24 • Baseline up to Week 100

Additional Information

Medical Director

Sarepta Therapeutics, Inc.

Phone: +1-888-727-3782

Results disclosure agreements

  • Principal investigator is a sponsor employee The most restrictive relevant agreement provides that the PI can only publish the study results with the approval of Sponsor.
  • Publication restrictions are in place

Restriction type: OTHER