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

3 de setembro de 2026 atualizado por: 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.

Visão geral do estudo

Descrição detalhada

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?

Tipo de estudo

Observacional

Inscrição (Estimado)

1000

Contactos e Locais

Esta seção fornece os detalhes de contato para aqueles que conduzem o estudo e informações sobre onde este estudo está sendo realizado.

Contato de estudo

Estude backup de contato

Locais de estudo

    • Virginia
      • Richmond, Virginia, Estados Unidos, 23298
        • Recrutamento
        • Virginia Commonwealth University
        • Investigador principal:
          • Nicholas Johnson, MD
        • Contato:

Critérios de participação

Os pesquisadores procuram pessoas que se encaixem em uma determinada descrição, chamada de critérios de elegibilidade. Alguns exemplos desses critérios são a condição geral de saúde de uma pessoa ou tratamentos anteriores.

Critérios de elegibilidade

Idades elegíveis para estudo

  • Adulto
  • Adulto mais velho

Aceita Voluntários Saudáveis

Não

Método de amostragem

Amostra Não Probabilística

População do estudo

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.

Descrição

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.

Plano de estudo

Esta seção fornece detalhes do plano de estudo, incluindo como o estudo é projetado e o que o estudo está medindo.

Como o estudo é projetado?

Detalhes do projeto

Coortes e Intervenções

Grupo / Coorte
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.

O que o estudo está medindo?

Medidas de resultados primários

Medida de resultado
Descrição da medida
Prazo
Characterize the long-term disease progression- 10 meter walk/run
Prazo: 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
Prazo: 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
Prazo: 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
Prazo: 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
Prazo: 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
Prazo: 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

Colaboradores e Investigadores

É aqui que você encontrará pessoas e organizações envolvidas com este estudo.

Investigadores

  • Investigador principal: Nicholas Johnson, MD, Virginia Commonwealth University

Publicações e links úteis

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Publicações Gerais

Datas de registro do estudo

Essas datas acompanham o progresso do registro do estudo e os envios de resumo dos resultados para ClinicalTrials.gov. Os registros do estudo e os resultados relatados são revisados ​​pela National Library of Medicine (NLM) para garantir que atendam aos padrões específicos de controle de qualidade antes de serem publicados no site público.

Datas Principais do Estudo

Início do estudo (Estimado)

1 de setembro de 2026

Conclusão Primária (Estimado)

1 de dezembro de 2032

Conclusão do estudo (Estimado)

1 de dezembro de 2032

Datas de inscrição no estudo

Enviado pela primeira vez

8 de julho de 2026

Enviado pela primeira vez que atendeu aos critérios de CQ

8 de julho de 2026

Primeira postagem (Real)

13 de julho de 2026

Atualizações de registro de estudo

Última Atualização Postada (Real)

4 de setembro de 2026

Última atualização enviada que atendeu aos critérios de controle de qualidade

3 de setembro de 2026

Última verificação

1 de setembro de 2026

Mais Informações

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