- ICH GCP
- Registro degli studi clinici negli Stati Uniti
- Sperimentazione clinica NCT07700225
Establishing Biomarkers and Clinical Endpoints in Myotonic Dystrophy Type 1 (END-DM1) Extension (END-EXT)
8 luglio 2026 aggiornato da: 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.
Panoramica dello studio
Stato
Reclutamento
Descrizione dettagliata
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:
- How do clinical measures, such as walking speed, hand function, and muscle strength, change over a multi-year period in people with DM1?
- 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 di studio
Osservativo
Iscrizione (Stimato)
1000
Contatti e Sedi
Questa sezione fornisce i recapiti di coloro che conducono lo studio e informazioni su dove viene condotto lo studio.
Contatto studio
- Nome: Jennifer Raymond
- Numero di telefono: 804-828-6318
- Email: Jennifer.raymond@vcuhealth.org
Backup dei contatti dello studio
- Nome: Ruby Langeslay
- Numero di telefono: 804-828-6318
- Email: Ruby.langeslay@vcuhealth.org
Luoghi di studio
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Virginia
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Richmond, Virginia, Stati Uniti, 23298
- Reclutamento
- Virginia Commonwealth University
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Investigatore principale:
- Nicholas Johnson, MD
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Contatto:
- Jennifer Raymond
- Numero di telefono: 804-828-6318
- Email: Jennifer.raymond@vcuhealth.org
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Criteri di partecipazione
I ricercatori cercano persone che corrispondano a una certa descrizione, chiamata criteri di ammissibilità. Alcuni esempi di questi criteri sono le condizioni generali di salute di una persona o trattamenti precedenti.
Criteri di ammissibilità
Età idonea allo studio
- Adulto
- Adulto più anziano
Accetta volontari sani
No
Metodo di campionamento
Campione non probabilistico
Popolazione di studio
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.
Descrizione
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.
Piano di studio
Questa sezione fornisce i dettagli del piano di studio, compreso il modo in cui lo studio è progettato e ciò che lo studio sta misurando.
Come è strutturato lo studio?
Dettagli di progettazione
Coorti e interventi
Gruppo / Coorte |
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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.
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Cosa sta misurando lo studio?
Misure di risultato primarie
Misura del risultato |
Misura Descrizione |
Lasso di tempo |
|---|---|---|
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Characterize the long-term disease progression- 10 meter walk/run
Lasso di tempo: Baseline (0 months), every 12 months over four years
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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.
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Baseline (0 months), every 12 months over four years
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Characterize the long-term disease progression- vHOT
Lasso di tempo: Baseline (0 months), every 12 months over four years
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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.
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Baseline (0 months), every 12 months over four years
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Characterize the long-term disease progression- grip strength
Lasso di tempo: Baseline (0 months), every 12 months over four years
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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).
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Baseline (0 months), every 12 months over four years
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Characterize the long-term disease progression- ECG
Lasso di tempo: Baseline (0 months), every 12 months over four years
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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.
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Baseline (0 months), every 12 months over four years
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Characterize the long-term disease progression- FVC
Lasso di tempo: Baseline (0 months), every 12 months over four years
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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.
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Baseline (0 months), every 12 months over four years
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Characterize the long-term disease progression- DM1-Activ-c
Lasso di tempo: Baseline (0 months), every 12 months over four years
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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.
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Baseline (0 months), every 12 months over four years
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Collaboratori e investigatori
Qui è dove troverai le persone e le organizzazioni coinvolte in questo studio.
Sponsor
Investigatori
- Investigatore principale: Nicholas Johnson, MD, Virginia Commonwealth University
Pubblicazioni e link utili
La persona responsabile dell'inserimento delle informazioni sullo studio fornisce volontariamente queste pubblicazioni. Questi possono riguardare qualsiasi cosa relativa allo studio.
Pubblicazioni generali
- Guyatt GH, Osoba D, Wu AW, Wyrwich KW, Norman GR; Clinical Significance Consensus Meeting Group. Methods to explain the clinical significance of health status measures. Mayo Clin Proc. 2002 Apr;77(4):371-83. doi: 10.4065/77.4.371.
- Jaeschke R, Singer J, Guyatt GH. Measurement of health status. Ascertaining the minimal clinically important difference. Control Clin Trials. 1989 Dec;10(4):407-15. doi: 10.1016/0197-2456(89)90005-6.
- Mathai SC, Puhan MA, Lam D, Wise RA. The minimal important difference in the 6-minute walk test for patients with pulmonary arterial hypertension. Am J Respir Crit Care Med. 2012 Sep 1;186(5):428-33. doi: 10.1164/rccm.201203-0480OC. Epub 2012 Jun 21.
- Goldman A, Ramsay M, Jenkins T. Ethnicity and myotonic dystrophy: a possible explanation for its absence in sub-Saharan Africa. Ann Hum Genet. 1996 Jan;60(1):57-65. doi: 10.1111/j.1469-1809.1996.tb01172.x.
- Griggs RC, Wood DS. Criteria for establishing the validity of genetic recombination in myotonic dystrophy. Neurology. 1989 Mar;39(3):420-1. doi: 10.1212/wnl.39.3.420. No abstract available.
- Personius KE, Pandya S, King WM, Tawil R, McDermott MP. Facioscapulohumeral dystrophy natural history study: standardization of testing procedures and reliability of measurements. The FSH DY Group. Phys Ther. 1994 Mar;74(3):253-63. doi: 10.1093/ptj/74.3.253.
- Thornton CA, Johnson K, Moxley RT 3rd. Myotonic dystrophy patients have larger CTG expansions in skeletal muscle than in leukocytes. Ann Neurol. 1994 Jan;35(1):104-7. doi: 10.1002/ana.410350116.
- Braida C, Stefanatos RK, Adam B, Mahajan N, Smeets HJ, Niel F, Goizet C, Arveiler B, Koenig M, Lagier-Tourenne C, Mandel JL, Faber CG, de Die-Smulders CE, Spaans F, Monckton DG. Variant CCG and GGC repeats within the CTG expansion dramatically modify mutational dynamics and likely contribute toward unusual symptoms in some myotonic dystrophy type 1 patients. Hum Mol Genet. 2010 Apr 15;19(8):1399-412. doi: 10.1093/hmg/ddq015. Epub 2010 Jan 15.
- Guyatt G, Walter S, Norman G. Measuring change over time: assessing the usefulness of evaluative instruments. J Chronic Dis. 1987;40(2):171-8. doi: 10.1016/0021-9681(87)90069-5.
- Wyrwich KW, Tierney WM, Wolinsky FD. Further evidence supporting an SEM-based criterion for identifying meaningful intra-individual changes in health-related quality of life. J Clin Epidemiol. 1999 Sep;52(9):861-73. doi: 10.1016/s0895-4356(99)00071-2.
- Teng S, Wang B, Yang F, Yi X, Zhang X, Sun Y. MediDRNet: Tackling category imbalance in diabetic retinopathy classification with dual-branch learning and prototypical contrastive learning. Comput Methods Programs Biomed. 2024 Aug;253:108230. doi: 10.1016/j.cmpb.2024.108230. Epub 2024 May 17.
- Liu P, Liu Y, Liu H, Xiong L, Mei C, Yuan L. A Random Forest Algorithm for Assessing Risk Factors Associated With Chronic Kidney Disease: Observational Study. Asian Pac Isl Nurs J. 2024 Jun 3;8:e48378. doi: 10.2196/48378.
Studiare le date dei record
Queste date tengono traccia dell'avanzamento della registrazione dello studio e dell'invio dei risultati di sintesi a ClinicalTrials.gov. I record degli studi e i risultati riportati vengono esaminati dalla National Library of Medicine (NLM) per assicurarsi che soddisfino specifici standard di controllo della qualità prima di essere pubblicati sul sito Web pubblico.
Studia le date principali
Inizio studio (Stimato)
1 luglio 2026
Completamento primario (Stimato)
1 dicembre 2032
Completamento dello studio (Stimato)
1 dicembre 2032
Date di iscrizione allo studio
Primo inviato
8 luglio 2026
Primo inviato che soddisfa i criteri di controllo qualità
8 luglio 2026
Primo Inserito (Effettivo)
13 luglio 2026
Aggiornamenti dei record di studio
Ultimo aggiornamento pubblicato (Effettivo)
13 luglio 2026
Ultimo aggiornamento inviato che soddisfa i criteri QC
8 luglio 2026
Ultimo verificato
1 luglio 2026
Maggiori informazioni
Termini relativi a questo studio
Parole chiave
Termini MeSH pertinenti aggiuntivi
- Manifestazioni neurologiche
- Malattie muscoloscheletriche
- Malattie del sistema nervoso
- Malattie muscolari
- Manifestazioni neuromuscolari
- Malattie genetiche, congenite
- Malattie Neurodegenerative
- Malattie Eredodegenerative, Sistema Nervoso
- Disturbi muscolari, atrofico
- Disturbi miotonici
- Malattie e anomalie congenite, ereditarie e neonatali
- Condizioni patologiche, segni e sintomi
- Segni e sintomi
- Distrofie muscolari
- Distrofia miotonica
- Miotonia
- Malattie neuromuscolari
Altri numeri di identificazione dello studio
- HM300000528 END-EXT
Informazioni su farmaci e dispositivi, documenti di studio
Studia un prodotto farmaceutico regolamentato dalla FDA degli Stati Uniti
No
Studia un dispositivo regolamentato dalla FDA degli Stati Uniti
No
Queste informazioni sono state recuperate direttamente dal sito web clinicaltrials.gov senza alcuna modifica. In caso di richieste di modifica, rimozione o aggiornamento dei dettagli dello studio, contattare register@clinicaltrials.gov. Non appena verrà implementata una modifica su clinicaltrials.gov, questa verrà aggiornata automaticamente anche sul nostro sito web .
Prove cliniche su Distrofia miotonica di tipo 1
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Assistance Publique - Hôpitaux de ParisURC Necker Cochin, FranceReclutamentoArtrite idiopatica giovanile (AIG)Francia