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Establishing Biomarkers and Clinical Endpoints in Myotonic Dystrophy Type 1 (END-DM1) Extension (END-EXT)

3 september 2026 uppdaterad av: Virginia Commonwealth University
Myotonic Dystrophy type 1 (DM1) is an autosomal dominant multisystemic disorder that causes progressive disability and shortened life expectancy. It is characterized by progressive weakness and myotonia, which preferentially affects the craniofacial, hand, and distal leg muscles. Many patients also experience difficulties with cognition, cardiac arrhythmias, respiratory failure, or cataracts. Currently there is no treatment to slow progression or reverse the symptoms.

Studieöversikt

Detaljerad beskrivning

The goal of this observational study is to characterize long-term disease progression over at least 4 years in at least 1,000 adults with myotonic dystrophy type 1 (DM1).

The main questions this study aims to answer are:

  1. How do clinical measures, such as walking speed, hand function, and muscle strength, change over a multi-year period in people with DM1?
  2. Can long-term changes in slowly progressive measures, like heart rhythms (ECG) and lung function (FVC), be accurately captured and used as biomarkers for the disease over time?

Studietyp

Observationell

Inskrivning (Beräknad)

1000

Kontakter och platser

Det här avsnittet innehåller kontaktuppgifter för dem som genomför studien och information om var denna studie genomförs.

Studiekontakt

Studera Kontakt Backup

Studieorter

    • Virginia
      • Richmond, Virginia, Förenta staterna, 23298
        • Rekrytering
        • Virginia Commonwealth University
        • Huvudutredare:
          • Nicholas Johnson, MD
        • Kontakt:

Deltagandekriterier

Forskare letar efter personer som passar en viss beskrivning, så kallade behörighetskriterier. Några exempel på dessa kriterier är en persons allmänna hälsotillstånd eller tidigare behandlingar.

Urvalskriterier

Åldrar som är berättigade till studier

  • Vuxen
  • Äldre vuxen

Tar emot friska volontärer

Nej

Testmetod

Icke-sannolikhetsprov

Studera befolkning

The study seeks to enroll at least 1000 men and women aged 18 to 70 with a positive genetic test for myotonic dystrophy type 1, a clinical diagnosis of myotonic dystrophy type 1, or a clinical diagnosis or positive genetic test for congenital myotonic dystrophy. based on research criteria. Few restrictions are placed on participation in the study because we aim to capture the full spectrum of disease severity.

Beskrivning

Inclusion Criteria:

  • Age 18 to 70 years (inclusive)
  • Written, voluntary informed consent must be obtained prior to any study procedures. In cases where a Legally Authorized Representative (LAR) provides consent, verbal assent will be obtained from the subject, as determined by the investigator and documented directly on the consent form. Capacity to consent will be determined by the neurologist at the Baseline visit and will be signed off on the Inclusion/Exclusion checklist.
  • Clinical diagnosis of DM1 based on research criteria or positive genetic test. The research criteria for clinical diagnosis of DM1 require myotonia, muscle weakness in a characteristic distribution, and history of similar findings in a first degree relative. Genetic testing confirmed the diagnosis of DM1 in > 99% of individuals who satisfied these criteria. OR A diagnosis of CDM, which is defined as children having symptoms of myotonic dystrophy in the newborn period (<30 days), such as hypotonia, feeding or respiratory difficulty, requiring hospitalization to a ward or to the neonatal intensive care unit for 3 days or more; and a genetic test suspicious of an expanded trinucleotide (CTG) repeat in the DMPK gene in the child or first degree relative. An expanded CTG repeat size in the child is considered greater than 200 repeats or E1-E4 classification (E1= 200-500, E2=500-1,000, E3=1,000-1,500, E4>1,500)

Exclusion Criteria:

  • Symptomatic renal or liver disease, uncontrolled diabetes or thyroid disorder, or active malignancy other than in situ skin cancer.
  • Current alcohol or substance use disorder.
  • Concurrent pregnancy or planned pregnancy during the course of the study.
  • Concurrent medical condition that would, in the opinion of the investigator or clinical evaluator. compromise performance on study measures.
  • Use of mexiletine or other anti-myotonia agents within 72 hours of any study visit.

Studieplan

Det här avsnittet ger detaljer om studieplanen, inklusive hur studien är utformad och vad studien mäter.

Hur är studien utformad?

Designdetaljer

Kohorter och interventioner

Grupp / Kohort
Myotonic Dystrophy Type 1 (DM1) Longitudinal Cohort
Participants with a clinical or genetic diagnosis of myotonic dystrophy type 1 (DM1) or congenital myotonic dystrophy (CDM). This cohort includes individuals transitioning from the parent study, Establishing Biomarkers and Clinical Endpoints in Myotonic Dystrophy Type 1 (END-DM1; NCT03981575), as well as newly enrolled participants.

Vad mäter studien?

Primära resultatmått

Resultatmått
Åtgärdsbeskrivning
Tidsram
Characterize the long-term disease progression- 10 meter walk/run
Tidsram: Baseline (0 months), every 12 months over four years
The 10-meter walk test (10MWT) is used in research to reliably measure gait speed, assessing functional mobility and detecting changes in walking performance over time. It is a highly reliable, quick, and cost-effective tool. . It is favored for its simplicity, speed, and ability to predict functional independence. A "good" score varies by age/condition, with healthy adults averaging over 1.2-1.4 m/s.
Baseline (0 months), every 12 months over four years
Characterize the long-term disease progression- vHOT
Tidsram: Baseline (0 months), every 12 months over four years
The Video Hand Opening Time (vHOT) is used in research as a practical, low-cost, and reliable quantitative tool to measure handgrip myotonia (delayed muscle relaxation) in Myotonic Dystrophy Type 1 (DM1) patients. It is particularly valuable for multicenter clinical trials because it allows for blinded, objective assessment of therapeutic responses. A "good" (healthy) score is generally as close to zero as possible.
Baseline (0 months), every 12 months over four years
Characterize the long-term disease progression- grip strength
Tidsram: Baseline (0 months), every 12 months over four years
Grip strength is used in research as a reliable, low-cost biomarker for overall muscle strength, aging, and mortality risk. A good score varies by age and sex, with healthy young adults often averaging over 40-45 kg (males) and 25-30kg (females).
Baseline (0 months), every 12 months over four years
Characterize the long-term disease progression- ECG
Tidsram: Baseline (0 months), every 12 months over four years
Electrocardiograms (ECGs/EKGs) are used in research for their non-invasive, cost-effective ability to track heart rhythm, diagnose cardiac conditions, and assess cardiovascular disease risk. In research, a "good" ECG score indicates normal sinus rhythm and intervals, such as a PR interval of 120-200 milliseconds and an RR interval of 0.6-1.2 seconds.
Baseline (0 months), every 12 months over four years
Characterize the long-term disease progression- FVC
Tidsram: Baseline (0 months), every 12 months over four years
Forced Vital Capacity (FVC) is crucial in research for diagnosing and tracking restrictive lung diseases (e.g., pulmonary fibrosis), assessing disease progression, and measuring treatment efficacy in clinical trials. A "good" or normal FVC is typically 80% or higher of the predicted value, based on a patient's age, height, sex, and ethnicity.
Baseline (0 months), every 12 months over four years
Characterize the long-term disease progression- DM1-Activ-c
Tidsram: Baseline (0 months), every 12 months over four years
The DM1-Activ-c (25-item) is a validated, Rasch-built, patient-reported outcome measure designed specifically to assess daily activity and participation in Myotonic Dystrophy Type 1 (DM1) patients. It is used in research for its high sensitivity to disease progression and therapeutic changes (responsiveness), making it a reliable primary endpoint for clinical trials to measure patient improvement. The DM1-Activ-c is scored on a scale from 0 to 100, where higher scores indicate better functional ability.
Baseline (0 months), every 12 months over four years

Samarbetspartners och utredare

Det är här du hittar personer och organisationer som är involverade i denna studie.

Utredare

  • Huvudutredare: Nicholas Johnson, MD, Virginia Commonwealth University

Publikationer och användbara länkar

Den som ansvarar för att lägga in information om studien tillhandahåller frivilligt dessa publikationer. Dessa kan handla om allt som har med studien att göra.

Allmänna publikationer

Studieavstämningsdatum

Dessa datum spårar framstegen för inlämningar av studieposter och sammanfattande resultat till ClinicalTrials.gov. Studieposter och rapporterade resultat granskas av National Library of Medicine (NLM) för att säkerställa att de uppfyller specifika kvalitetskontrollstandarder innan de publiceras på den offentliga webbplatsen.

Studera stora datum

Studiestart (Beräknad)

1 september 2026

Primärt slutförande (Beräknad)

1 december 2032

Avslutad studie (Beräknad)

1 december 2032

Studieregistreringsdatum

Först inskickad

8 juli 2026

Först inskickad som uppfyllde QC-kriterierna

8 juli 2026

Första postat (Faktisk)

13 juli 2026

Uppdateringar av studier

Senaste uppdatering publicerad (Faktisk)

4 september 2026

Senaste inskickade uppdateringen som uppfyllde QC-kriterierna

3 september 2026

Senast verifierad

1 september 2026

Mer information

Denna information hämtades direkt från webbplatsen clinicaltrials.gov utan några ändringar. Om du har några önskemål om att ändra, ta bort eller uppdatera dina studieuppgifter, vänligen kontakta register@clinicaltrials.gov. Så snart en ändring har implementerats på clinicaltrials.gov, kommer denna att uppdateras automatiskt även på vår webbplats .

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