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Phase 3 N=12 Randomized Quadruple-blind Treatment

A Trial to Evaluate Safety, Tolerability and Efficacy of Elamipretide in Subjects With Barth Syndrome

Barth Syndrome

Enrolled (actual)
12
Serious AEs
14.7%
Results posted
Apr 2024
Primary outcome: Primary: Part 1: Distance Walked During the 6-Minute Walk Test (6MWT) by Visit — 400.1; 412.6; 443.1; 443.9 meters

Study Design & Population

Study type
Interventional
Phase
Phase 3
Interventions
Elamipretide (Drug); Placebo (Drug)
Age
Pediatric, Adult, Older Adult · 12+ yrs
Sex
Male
Sponsor
Stealth BioTherapeutics Inc.
Primary completion
Oct 2018

Outcome Measures

OutcomeResultp-value
PRIMARY
Part 1: Distance Walked During the 6-Minute Walk Test (6MWT) by Visit
400.1; 412.6; 443.1; 443.9
PRIMARY
Part 1: Total Fatigue Score Based on the BTHS-SA by Visit.
7.672; 7.433; 6.837; 6.994; 6.220; 6.485
SECONDARY
Part 2: Distance Walked During the 6MWT
45.4; 75.6; 79.8; 100.4; 68.0; 112.6
SECONDARY
Part 2: Change From Baseline: Total Fatigue Score (Q1, Q2, and Q4) Based on the BTHS-SA by Visit
-2.425; -1.540; -1.800; -1.314; -1.500; -0.314
SECONDARY
Part 1: Muscle Strength as Measured by HHD by Visit
131.236; 123.100; 135.934; 129.253
SECONDARY
Part 2: Change From Baseline: Muscle Strength by HHD (Newtons) by Visit
33.606; 42.147; 54.073; 54.068; 68.798; 48.351
SECONDARY
Part 1: 5XSST by Visit
12.965; 13.710; 14.023; 13.749
SECONDARY
Part 2: Change From Baseline: 5X Sit to Stand by Visit
-0.773; 0.400; -1.505; 0.384; -2.533; -1.160
SECONDARY
Part 1: SWAY Application Balance Assessments by Visit
70.752; 68.820; 78.698; 71.353
SECONDARY
Part 2: Change From Baseline: SWAY Application Balance Assessment
-3.152; 10.464; 12.190; 6.664; 0.807; 9.484
SECONDARY
Part 1: Patient Global Impression Scales of Symptoms
1.7; 1.8; 1.7; 1.5; 1.4; 1.6
SECONDARY
Part 2: Change From Baseline: Patient Global Impression Scales of Symptoms by Visit
0.0; -0.4; -0.2; -0.6; -0.3; -0.4
SECONDARY
Part 1: Part 1: Clinician Global Impression
1.8; 1.4; 1.6; 1.6
SECONDARY
Part 2: Change From Baseline: CGI Symptom Scale
-0.4; 0.0; -1.3; -0.4; -1.3; -0.2

Summary

A randomized, double-blind cross over trial to evaluate the safety, efficacy, and tolerability of elamipretide in subjects with Barth syndrome.

Eligibility Criteria

Inclusion Criteria

  • Genetically confirmed Barth Syndrome
  • Male aged 12 and above
  • At the screening visit, eGFR must meet the following:
  • Body weight >30 kg AND eGFR ≥ 90mL/min at screening
  • Body weight >40kg AND eGFR ≥60 but <90mL/min/ 1.73m² at screening
  • Ambulatory and impaired during the baseline 6MWT
  • On stable medication for 30 days prior to the baseline visit

Exclusion Criteria

  • Participated in another interventional clinical trial within 30 days of or is currently enrolled in a non-interventional clinical trial at the baseline visit potentially confounding with this trial
  • Prior or current medical condition that would prevent the subject from safely participating in the trial
  • Undergone any inpatient hospitalizations within 30 days of the baseline visit
  • Is undergoing an apparent pubertal growth spurt
  • Has uncontrolled hypertension
  • History of substance abused within the year before the baseline visit or is likely to be uncompliant
  • History of heart transplantation or current placement on the waiting list for a heart transplant
  • For subjects with an ICD: known occurrence of ICD discharge in the 3 months prior to the baseline visit
  • For subjects without an ICD: expected to undergo an implantation of an ICD during the conduct of the study
  • Currently receiving treatment with chemotherapeutic agents or immunosuppressant agents or has received prior radiation therapy to the chest
  • Recipient of stem cell or gene therapy or is currently being treated by a therapeutic investigational device
View full record on ClinicalTrials.gov →

Data sourced from ClinicalTrials.gov (NCT03098797). Outcome figures and adverse-event rates are extracted automatically from the registry's posted results and are provided for clinician reference, not as a substitute for the primary publication.

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