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

2026年9月3日 更新者: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.

研究概览

详细说明

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?

研究类型

观察性的

注册 (估计的)

1000

联系人和位置

本节提供了进行研究的人员的详细联系信息,以及有关进行该研究的地点的信息。

学习联系方式

研究联系人备份

学习地点

    • Virginia
      • Richmond、Virginia、美国、23298
        • 招聘中
        • Virginia Commonwealth University
        • 首席研究员:
          • Nicholas Johnson, MD
        • 接触:

参与标准

研究人员寻找符合特定描述的人,称为资格标准。这些标准的一些例子是一个人的一般健康状况或先前的治疗。

资格标准

适合学习的年龄

  • 成人
  • 年长者

接受健康志愿者

不

取样方法

非概率样本

研究人群

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.

描述

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.

学习计划

本节提供研究计划的详细信息,包括研究的设计方式和研究的衡量标准。

研究是如何设计的?

设计细节

队列和干预

团体/队列
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.

研究衡量的是什么?

主要结果指标

结果测量
措施说明
大体时间
Characterize the long-term disease progression- 10 meter walk/run
大体时间: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
大体时间: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
大体时间: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
大体时间: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
大体时间: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
大体时间: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

合作者和调查者

在这里您可以找到参与这项研究的人员和组织。

调查人员

  • 首席研究员:Nicholas Johnson, MD、Virginia Commonwealth University

出版物和有用的链接

负责输入研究信息的人员自愿提供这些出版物。这些可能与研究有关。

一般刊物

研究记录日期

这些日期跟踪向 ClinicalTrials.gov 提交研究记录和摘要结果的进度。研究记录和报告的结果由国家医学图书馆 (NLM) 审查,以确保它们在发布到公共网站之前符合特定的质量控制标准。

研究主要日期

学习开始 (估计的)

2026年9月1日

初级完成 (估计的)

2032年12月1日

研究完成 (估计的)

2032年12月1日

研究注册日期

首次提交

2026年7月8日

首先提交符合 QC 标准的

2026年7月8日

首次发布 (实际的)

2026年7月13日

研究记录更新

最后更新发布 (实际的)

2026年9月4日

上次提交的符合 QC 标准的更新

2026年9月3日

最后验证

2026年9月1日

更多信息

此信息直接从 clinicaltrials.gov 网站检索,没有任何更改。如果您有任何更改、删除或更新研究详细信息的请求,请联系 register@clinicaltrials.gov. clinicaltrials.gov 上实施更改,我们的网站上也会自动更新.

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