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Motor Outcomes to Validate Evaluations in FSHD (MOVE FSHD)

Recruiting
Conditions
FSHD
Registration Number
NCT04635891
Lead Sponsor
University of Kansas Medical Center
Brief Summary

The primary goal of this proposal is to collect motor and functional outcomes specific to FSHD over time. By collecting measures specific to FSHD, this will help ensure the best level of clinical care is being provided. Also, the hope is to speed up drug development by gaining a better understanding of how having FSHD impacts motor function and other health outcomes (i.e. breathing, wheelchair use, etc.) and how big a change in motor function would be clinically meaningful to those with FSHD.

Motor Outcomes to Validate Evaluations in FSHD (MOVE FSHD) will have approximately 450 FSHD participants followed for a minimum of 3 years. A subset of MOVE FSHD participants, approximately 200, will participate in the MOVE+ sub-study which includes whole body MRI and reachable workspace, as well as optional muscle biopsy and wearable device (US participants only).

Detailed Description

Not available

Recruitment & Eligibility

Status
RECRUITING
Sex
All
Target Recruitment
450
Inclusion Criteria
  • Genetically confirmed FSHD (types 1 or 2) or clinical diagnosis of FSHD with characteristic findings on exam and an affected parent or offspring.
Exclusion Criteria
  • Unwilling or unable to provide informed consent.
  • Any other medical condition which in the opinion of the investigator would interfere with study participation.

Study & Design

Study Type
OBSERVATIONAL
Study Design
Not specified
Primary Outcome Measures
NameTimeMethod
10m walk/runBaseline - 5 years

The 10-meter walk/run (previously the 30 foot go) or gait speed task will be performed during study visits. This task tests a range of different abilities, from power, to endurance, and balance. Also, the 10 meter walk/run is a predictor of loss of ambulation in Duchenne Muscular Dystrophy.

Shoulder and Arm Range of MotionBaseline - 5 years

Range of motion tasks mimicking lifting or reaching up will be performed.

Shoulder and Arm FunctionBaseline - 5 years

Participants will be timed on stacking cans.

Spirometry (FVC, FEV1, PCF)Baseline - 5 years

Investigators will obtain forced vital capacity and forced expiratory volume in 1 second, both standardized outcomes used commonly in clinic and clinical trials. Also, for sites who routinely collect Peak Cough Flow this will also be obtained.

Secondary Outcome Measures
NameTimeMethod
MIP/MEP and SNIPBaseline - 5 years

For sites who routinely obtain negative inspiratory or expiratory force (MIP/MEP) will be collected.

4 Stair ClimbBaseline - 5 years

Time to ascend and descend 4 standard stairs.

Manual Muscle Testing (MMT)Baseline - 5 years

For MMT a modified Medical Research Council 13-point scale will be used - with standardized positions for each muscle.

Exercise and Pain AssessmentBaseline - 5 years

An exercise and pain assessment questionnaire was comprised specifically for this study and will be completed at every visit.

The Facial Disability Index (FDI)Baseline - 5 years

The FDI is a short 5 item questionnaire. The five questions are summed into total score which transformed onto a percentage scale, with 100 representing normal, and lower scores representing increasing disability.

Trunk FunctionBaseline - 5 years

There are no specific functional tasks designed to measure trunk function in FSHD so Investigators have chosen a practical task that will reflect changes in core truncal muscle groups. The ability to sit up and the timed supine to sitting test reflects core muscle strength and coordination.

Hand FunctionBaseline - 5 years

Hand function is captured by examining hand grip strength.

Timed Up and Go (TUG)Baseline - 5 years

Balance and mobility are assessed by using the Timed Up and Go (TUG), a standard outcome measure for the elderly that is also increasingly being used in neuromuscular disorders. Participants are asked to rise from a chair, walk 3 meters, turn 180 degrees and return to a seated position in the chair.

Saliva MethylationBaseline (collected only once)

Investigators will isolate DNA from blood cells in saliva - and will determine the methylation levels in the 4q region on chromosome 4 using this new technique. Investigators will compare methylation levels between participants and compare to other clinical information collected in this study.

FSHD Clinical Severity ScoresBaseline - 5 years

A limited physical exam and strength testing will be used to derive two FSHD clinical severity scores. These severity scores both rank weakness in the face, shoulders, arms, distal, and proximal lower extremities on either a 10 or 15 point scale. The higher the severity score the more affected the individual.

Patient-Reported Outcomes Measurement Information System-57 (PROMIS57)Baseline - 5 years

The PROMIS57 is an instrument developed by the NIH which generates scores for physical function, and the impact of physical limitations on daily life. 57 questions are summed into a total score, which is transformed into a normalized t-score with 50 representing normal, and lower scores representing increasing disability.

The Upper Extremity Functional IndexBaseline - 5 years

This index measures upper extremity dysfunction. 20 questions are combined into a total score, the score is transformed into a normalized score with 80 representing normal, and lower scores representing increasing disability.

FSHD Rasch-built overall disability scales (FSHD-RODS)Baseline - 5 years

The FSHD-RODS is a questionnaire about the relationship between daily activities and health. 32 questions are are constructed using a modern clinimetric technique, the Rasch analysis, which by ordering the items in increasing difficulty, allows for the calculation of the sum of item scores to achieve a total score. Answers provide information about how FSHD affects daily and social activities and to what degree participants are able to perform usual activities.

Trial Locations

Locations (20)

Austin Neuromuscular Center

🇺🇸

Austin, Texas, United States

University of Texas Health San Antonio

🇺🇸

San Antonio, Texas, United States

University of Sao Paulo

🇧🇷

Sao Paulo, Brazil

David Geffen School of Medicine at UCLA

🇺🇸

Los Angeles, California, United States

Neuromuscular Disorders Program at Stanford University School of Medicine

🇺🇸

Stanford, California, United States

University of Colorado Anschutz Medical Campus

🇺🇸

Aurora, Colorado, United States

Univeristy of Florida Gainesville

🇺🇸

Gainesville, Florida, United States

University of Iowa

🇺🇸

Iowa City, Iowa, United States

Univeristy of Kansas Medical Center

🇺🇸

Kansas City, Kansas, United States

University of Calgary

🇨🇦

Calgary, Alberta, Canada

Kennedy Krieger Institute

🇺🇸

Baltimore, Maryland, United States

University of Rochester Medical Center

🇺🇸

Rochester, New York, United States

The Ohio State University Medical Center

🇺🇸

Columbus, Ohio, United States

Univeristy of Texas Southwestern Medical Center

🇺🇸

Dallas, Texas, United States

University of Utah

🇺🇸

Salt Lake City, Utah, United States

Virginia Commonwealth University

🇺🇸

Richmond, Virginia, United States

University of Washington Medical Center

🇺🇸

Seattle, Washington, United States

Ottawa Hospital Research Institute

🇨🇦

Ottawa, Ontario, Canada

University of McGill

🇨🇦

Montreal, Quebec, Canada

Sheffield Teaching Hospital

🇬🇧

Sheffield, South Yorkshire, United Kingdom

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