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Zebrafish dspb-/- mutant as a model for non-dilated left ventricular cardiomyopathy: exploring cardiac dysfunction
Serena Munteanu1, Jesús Wagih Gómez1, Ángel Júdez Serrano2
1Cardiogenetic Laboratory, Biomedical Research Institute of Murcia - Pascual Parrilla (IMIB-PP), University of Murcia, Murcia, Spain.
Molecular Biomedicine
|June 15, 2026
Summary
Physical activity may modify non-dilated left ventricular cardiomyopathy (ND-LVC) in DSP variant carriers. Moderate exercise improved cardiac function and endurance in a zebrafish model without increasing mortality, suggesting lifestyle factors impact disease progression.
Area of Science:
- Cardiology
- Genetics
- Exercise Physiology
Background:
- Truncating DSP gene variants cause non-dilated left ventricular cardiomyopathy (ND-LVC), impairing heart function without dilation.
- The role of physical activity in ND-LVC onset, severity, and progression is not well understood.
Purpose of the Study:
- To create and characterize a zebrafish model of DSP-related ND-LVC.
- To investigate the impact of moderate and endurance exercise on cardiac function, structure, and survival in this model.
- To analyze clinical data from human DSP variant carriers regarding physical activity and disease outcomes.
Main Methods:
- Generated a CRISPR/Cas9-induced homozygous dspb-/- zebrafish model (p.T449fs*) to study cardiac phenotypes.
- Applied moderate exercise training protocols to assess effects on cardiac function, endurance, and survival.
- Analyzed clinical data from human carriers correlating physical activity with age at diagnosis, disease severity, and arrhythmias.
Main Results:
- Zebrafish mutants displayed baseline systolic dysfunction, sarcomeric disorganization, and dysregulated signaling pathways.
- Moderate exercise partially restored cardiac function and improved endurance in mutants without increasing mortality.
- Physically active human carriers were diagnosed earlier but showed no increased disease severity or arrhythmia rates.
Conclusions:
- Exercise may act as a phenotypic modifier in DSP-related ND-LVC, potentially improving functional outcomes.
- The dspb-/- zebrafish model serves as a valuable platform for studying lifestyle factor interactions in desmosomal cardiomyopathies.
- Moderate physical activity appears beneficial for DSP-related ND-LVC, enhancing function without accelerating disease progression.
