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CompletedNCT03351270NatHis-CNMUpdated Mar 2, 2023

Prospective Natural History Study of Patients With Myotubular Myopathy and Other CentroNuclear Myopathies

An interventional study of Standardized strength, respiratory and motor function assessments in Centronuclear Myopathy, sponsored by Institut de Myologie, France. Completed at 11 sites in 5 countries. Per ClinicalTrials.gov, last updated 2023-03-02.

Sponsored by Institut de Myologie, France · Not applicable, Interventional, and Other

Phase
Not applicable
Study type
Interventional
Enrollment
60
Allocation
Not applicable
Sex
All
01

Study summary

This is the 2 years extension of the prospective and longitudinal study of the natural history and functional status of patients with myotubular myopathy and other centronuclear (CNM) sponsored by Dynacure including ten additional pediatric patients with mutation in MTM1 or DNM2 genes). the patients are planned to be enrolled in one year leading to an expected total number of 70 patients followed at least over 1 year period. Data from the study will be used to characterize the disease course of CNM and determine which outcome measures will be the best to assess the efficacy of potential therapies.

02

Conditions studied

  • Centronuclear Myopathy

Keywords

  • CNM
  • XLMTM
  • Myotubular myopathy
03

Who can participate

Ages eligible
Child (0–17), Adult (18–64), Older adult (65+)
Sexes eligible
All
Accepts healthy volunteers
No

Inclusion criteria

  • Patients of any age (newborns included) may participate
  • Patients over 18 years of age and parent(s)/legal guardian(s) of patients \< 18 years of age must be provide written informed consent prior to participating in the study and informed assent will be obtained from minors at least 7 years of age when required by regulation.
  • Myotubular Centronuclear myopathy (MTMCNM) resulting from a mutation in the MTM1, DNM2 or BIN1 gene
  • Male or symptomatic female. A symptomatic female will be defined by the motor function assessment by Motor Function Measure (MFM) or North Star Ambulatory Assessment (NSAA) below 80% of the total score.
  • Willing and able to comply with all protocol requirements and procedures.
  • In France only: Affiliated to or a beneficiary of a social security category

Exclusion criteria

Exclusion Criteria:

  • Other disease which may significantly interfere with the assessment of the MTM CNM and is clearly not related to the disease
  • Currently enrolled in a treatment study; or treatment with an experimental therapy other than pyridostigmine
  • For women: pregnancy or current breastfeeding
04

Study design

Phase
Not applicable
Primary purpose
Other
Allocation
Not applicable
Intervention model
Single group
Masking
None (open label)
Enrollment
60 participants (actual)

Interventions

  • OtherStandardized strength, respiratory and motor function assessments

    Standardized assessments will be adjusted by the age, the ambulant status and the respiratory status of the patient. The visit frequency will also be adjusted according to the age and to the time spent in the study varying between quarterly to yearly.

05

What researchers measure

Primary outcomes

  1. MFM score change from baseline

    Motor Function Measure scale. The total test score can range from 0 if the subject cannot perform any of the items to 100 if all the items are fully achieved.

    Time frame: Baseline, 6 months, 12 months and every year up to 60 months

  2. CHOP-INTEND score change from baseline

    Children's Hospital of Philadelphia Infant Test of Neuromuscular Disorders

    Time frame: Baseline, 6 months, 12 months and every year up to 60 months

  3. Modified Hammersmith score change from baseline

    Modified Hammersmith Functional Motor Scale. The total test score can range from 0 if the subject cannot perform any of the items to 40 if all the items are fully achieved

    Time frame: Baseline, 6 months, 12 months and every year up to 60 months

  4. Moviplate score change from baseline

    Moviplate score

    Time frame: Baseline, 6 months, 12 months and every year up to 60 months

  5. 6MWD change from baseline

    6 Minute Walking Distance

    Time frame: Baseline, 6 months, 12 months and every year up to 60 months

  6. Grip strength change from baseline

    MyoGrip

    Time frame: Baseline, 6 months, 12 months and every year up to 60 months

  7. Pinch strength change from baseline

    MyoPinch

    Time frame: Baseline, 6 months, 12 months and every year up to 60 months

  8. Forced Vital Capacity change from baseline

    Time frame: Baseline, 6 months, 12 months and every year up to 60 months

  9. Peak Cough Flow change from baseline

    Time frame: Baseline, 6 months, 12 months and every year up to 60 months

  10. Maximum Inspiratory Pressure change from baseline

    Time frame: Baseline, 6 months, 12 months and every year up to 60 months

  11. Maximum Expiratory Pressure change from baseline

    Time frame: Baseline, 6 months, 12 months and every year up to 60 months

06

Study locations

11 sites
  • Centre de référence neuromusculaire, CHR La Citadelle
    Liège, 4000, Belgium
  • Hôpital Femme Mère Enfant, CHU Lyon L'Escale
    Bron, 69500, France
  • Hôpital Roger Salengro, CHU Lille
    Lille, 59000, France
  • Hôpital de la Croix Rousse
    Lyon, 69004, France
  • Hôpital Armand Trousseau
    Paris, 75012, France
  • I-Motion Institute - Trousseau Hospital
    Paris, 75012, France
  • Institute of Myology
    Paris, 75013, France
  • Hôpital Sainte Musse
    Toulon, 83056, France
  • Universitätsklinikum Essen (AöR)
    Essen, D-45147, Germany
  • Bambino Gesu Children's Hospital
    Roma, 00146, Italy
  • Hospital Puerta del Mar
    Cadiz, 21-11009, Spain
07

Registry details

Key details

Study ID
NCT03351270
Lead sponsor
Institut de Myologie, France
Collaborators
Dynacure
Responsible party
Sponsor
First posted
Nov 22, 2017
Start date
May 28, 2017
Primary completion
Sep 30, 2021
Completion
Sep 30, 2021
Last update
Mar 2, 2023

Study contacts

Laurent Servais, MD
principal investigator · Centre de référence neuromusculaire, CHR La Citadelle; 4000 Liege Belgium

Oversight

Data monitoring committee
No
FDA-regulated drug
No
FDA-regulated device
No
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