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A Phase 2b Study of Zagociguat in Patients With MELAS

Phase 2
Recruiting
Conditions
Mitochondrial Encephalopathy, Lactic Acidosis and Stroke-Like Episodes (MELAS Syndrome)
Interventions
Registration Number
NCT06402123
Lead Sponsor
Tisento Therapeutics
Brief Summary

PRIZM is a Phase 2b randomized, double-blind, placebo-controlled, 3-treatment, 2-period, crossover study evaluating the efficacy and safety of oral zagociguat 15 and 30 mg vs. placebo when administered daily for 12 weeks in participants with genetically and phenotypically defined MELAS.

Detailed Description

The goal of this clinical trial is to learn about the effectiveness of zagociguat in patients with MELAS. The main questions it aims to answer are:

* Does zagociguat improve fatigue in patients with MELAS?

* Does zagociguat improve cognitive performance in patients with MELAS?

* What is the safety and tolerability profile of zagociguat?

The PRIZM study is evaluating 2 dose levels of zagociguat in a crossover design consisting of two 12-week treatment periods separated by a 4-week washout. Patients will be screened and if eligible, randomly assigned either to receive placebo in period 1 followed by active drug in period 2 OR to receive active drug in period 1 followed by placebo in period 2. Study medication is a once daily oral tablet and will be provided at the clinic and/or shipped to the participant's home. Clinic visits will occur at screening and Week 1 and Week 12 of each treatment period. Visits at Week 4 and Week 8 of both crossover periods will either be in clinic or optionally at the participant's home. Study assessments will be conducted weekly on a phone app and a separate tablet and additional assessments will be conducted during visits. Patients who complete the study will be eligible for an open label extension study.

Recruitment & Eligibility

Status
RECRUITING
Sex
All
Target Recruitment
44
Inclusion Criteria
  1. Signed consent form.

  2. 18 to 75 years of age.

  3. Diagnosed with MELAS based on the presence of each of the following criteria:

    1. A documented pathogenic variant in a mitochondrial DNA (mtDNA) gene.
    2. History of one or more stroke-like episodes (SLEs) with magnetic resonance imaging (MRI) findings consistent with stroke-like lesions.
  4. Scores below normal average on the iDSST and GMLT.

  5. Reports fatigue due to MELAS.

  6. Can complete at least 1 sit-to-stand in the 30-second test interval.

  7. Completes all at-home weekly activities independently during the Screening Period (caregiver may help set up device/app, log-in, etc.).

  8. Other criteria per the protocol.

Exclusion Criteria
  1. Systolic blood pressure (BP) 90 mmHg or diastolic BP 60 mmHg.
  2. Orthostatic hypotension when measured after standing from a semi-recumbent/supine position.
  3. Active cancer significant enough to confound the results of this study.
  4. Severe gastrointestinal dysmotility that may impact participation.
  5. Recent history (within last 6 months) of platelet dysfunction, hemophilia, von Willebrand disease, coagulation disorder, other bleeding diathesis condition(s), or significant, nontraumatic bleeding episodes.
  6. History of spontaneous fracture(s) that in the investigator's opinion represents a safety risk for trial participation.
  7. Current use of prohibited medication (reviewed by investigator).
  8. Any medical or other condition that the investigator thinks would preclude study participation.
  9. Other exclusion criteria per protocol.

Study & Design

Study Type
INTERVENTIONAL
Study Design
CROSSOVER
Arm && Interventions
GroupInterventionDescription
Placebo then Zagociguat 15mgzagociguat 15mgParticipants first receive placebo once a day for 12 weeks. Then after a 4 week washout, they receive zagociguat 15 mg once a day for 12 weeks.
Placebo then Zagociguat 15mgPlaceboParticipants first receive placebo once a day for 12 weeks. Then after a 4 week washout, they receive zagociguat 15 mg once a day for 12 weeks.
Zagociguat 15mg then PlaceboPlaceboParticipants first receive zagociguat 15 mg once a day for 12 weeks. Then after a 4-week washout, they receive placebo once a day for 12 weeks.
Placebo then Zagociguat 30mgPlaceboParticipants first receive placebo once a day for 12 weeks. Then after a 4-week washout, they receive zagociguat 30 mg once a day for 12 weeks.
Zagociguat 30mg then PlaceboPlaceboParticipants first receive zagociguat 30 mg once a day for 12 weeks. Then after a 4-week washout, they receive placebo once a day for 12 weeks.
Zagociguat 15mg then Placebozagociguat 15mgParticipants first receive zagociguat 15 mg once a day for 12 weeks. Then after a 4-week washout, they receive placebo once a day for 12 weeks.
Placebo then Zagociguat 30mgzagociguat 30mgParticipants first receive placebo once a day for 12 weeks. Then after a 4-week washout, they receive zagociguat 30 mg once a day for 12 weeks.
Zagociguat 30mg then Placebozagociguat 30mgParticipants first receive zagociguat 30 mg once a day for 12 weeks. Then after a 4-week washout, they receive placebo once a day for 12 weeks.
Primary Outcome Measures
NameTimeMethod
• PROMIS Fatigue MELAS Short Form scores• Groton Maze Learning Test scores • International Digit Symbol Substitution Test scoresWeeks 9 through 12 of each treatment period

These measures are a patient reported questionnaire on MELAS-specific fatigue and 2 cognitive performance tests. These 3 outcome measures will be combined using a global statistical test.

Incidence of Treatment-emergend Adverse Events (TEAEs)Weeks 1 through washout for treatment period 1 and Weeks 1 through follow-up for treatment period 2

TEAEs are any untoward event that may or may not be related to study medication.

Secondary Outcome Measures
NameTimeMethod
Plasma concentrations of GDF-15Week 12

Measure of disease pathophysiology

Number of repetitions completed during the 30-second sit-to-stand testWeek 12

Leg strength and exercise intolerance

PROMIS Cognitive Function MELAS Short Form (PCFM-SF) scoreWeek 12

Patient-reported questionnaire on MELAS-specific cognitive function

Memory composite scores (One Card Learning and One Back Tests) for Week 9 through 12Week 9 through 12

Test of memory

Trial Locations

Locations (25)

UC San Diego - Altman Clinical and Translational Research Institute

🇺🇸

La Jolla, California, United States

Children's Hospital of Colorado

🇺🇸

Aurora, Colorado, United States

Children's National

🇺🇸

Washington, District of Columbia, United States

Rare Disease Research

🇺🇸

Atlanta, Georgia, United States

Johns Hopkins University School of Medicine

🇺🇸

Baltimore, Maryland, United States

Massachusetts General Hospital

🇺🇸

Boston, Massachusetts, United States

Mayo Clinic Rochester

🇺🇸

Rochester, Minnesota, United States

Columbia University Irving Medical Center

🇺🇸

New York, New York, United States

Mount Sinai - Ichan School of Medicine

🇺🇸

New York, New York, United States

Akron Children's Hospital

🇺🇸

Akron, Ohio, United States

Children's Hospital of Philadelphia

🇺🇸

Philadelphia, Pennsylvania, United States

UPMC Children's Hospital of Pittsburgh

🇺🇸

Pittsburgh, Pennsylvania, United States

Baylor College of Medicine

🇺🇸

Houston, Texas, United States

University of Texas Medical School at Houston

🇺🇸

Houston, Texas, United States

Neuroscience Research Australia

🇦🇺

Sydney, New South Wales, Australia

Royal Melbourne Hospital

🇦🇺

Melbourne, Victoria, Australia

Shared Health/University of Manitoba

🇨🇦

Winnipeg, Manitoba, Canada

McMaster University Medical Center

🇨🇦

Hamilton, Ontario, Canada

University Hospital Bonn

🇩🇪

Bonn, Germany

Ludwig-Maximilians-University of Munich

🇩🇪

Munich, Germany

Neurologic Institute Carlo Besta of Milan

🇮🇹

Milan, Italy

University of Pisa Neurological Clinic

🇮🇹

Pisa, Italy

Fondazione Policlinico Universitario Agostino Gemelli

🇮🇹

Roma, Italy

UCL Queen Square Institute of Neurology

🇬🇧

London, United Kingdom

Newcastle University

🇬🇧

Newcastle Upon Tyne, United Kingdom

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