How the muscle protein MyBP-C controls skeletal muscle function
Skeletal Myosin-Binding Protein C: Defining Function Across Scales Using a Zebrafish Model System.
Researchers are using genetically engineered zebrafish to learn how changes in the MyBP-C protein lead to skeletal muscle problems like arthrogryposis.
Quick facts
| Grant type | R21 grant |
|---|---|
| Study type | NIH-funded research |
| Funding institution | University of Vermont & St Agric College NIH-funded |
| Lab location | 1 site (Burlington, United States) |
| Project ID | NIH-11182548 on NIH RePORTER |
What this research studies
This project makes engineered zebrafish muscles that express only one version of the myosin-binding protein C (MyBP-C) so scientists can see what each isoform does inside a working muscle. The team will use CRISPR/Cas9 GeneWeld to insert specific MyBP-C genes and then measure muscle contractility, actin-myosin interactions, and calcium sensitivity. Because mutations in human MYBPC1 and MYBPC2 are linked to conditions such as distal arthrogryposis, researchers will compare normal and mutant MyBP-C forms to connect molecular changes with muscle weakness or stiffness. Findings in zebrafish are intended to clarify disease mechanisms and point to targets for future treatments.
Who could benefit from this research
Good fit: People with genetic skeletal muscle disorders—especially those known to have MYBPC1 or MYBPC2 mutations or a diagnosis of distal arthrogryposis—would be most relevant to this research.
Not a fit: Patients whose muscle problems stem from unrelated injuries, metabolic causes, or non-MyBP-C genetic changes are unlikely to directly benefit from this project.
Why it matters
Potential benefit: If successful, this work could explain how specific MyBP-C mutations cause human skeletal muscle disease and point to targets for new therapies.
How similar studies have performed: Zebrafish and CRISPR methods have been used successfully to study muscle proteins before, but creating muscles that express only a single MyBP-C isoform is a novel application.
Where this research is happening
Burlington, United States
- University of Vermont & St Agric College — Burlington, United States (Active)
Researchers
- Principal investigator: Warshaw, David M — University of Vermont & St Agric College
- Study coordinator: Warshaw, David M
About this research
- This is an active NIH-funded research project — typically early-stage science, not a clinical trial accepting patient enrollment.
- Some NIH-funded labs run parallel clinical studies or seek volunteers for related work. To check, contact the principal investigator or institution listed above.
- For full project details, budget, and progress reports, visit the official NIH RePORTER page below.