Key result
A novel zebrafish Dmd-citrine gene trap line revealed that Dmd protein localizes specifically to the myosepta prior to dmd mRNA localization during muscle maturation.
The novel Dmd-citrine zebrafish line allows real-time tracking of dystrophin dynamics and serves as a model for assessing muscular dystrophy.
Supports dystrophin imaging in zebrafish models; leaves open translation to mammalian disease or clinical use.
Dystrophin (Dmd) is a structural protein that links the extracellular matrix to actin filaments in muscle fibers and is required for the maintenance of muscles integrity. Mutations in Dmd lead to muscular dystrophies in humans and other vertebrates. Here, we report the characterization of a zebrafish gene trap line that fluorescently labels the endogenous Dmd protein (Dmd-citrine, Gt(dmd-citrine) ct90a). We show that the Dmd-citrine line recapitulates endogenous dmd transcript expression and Dmd protein localization. Using this Dmd-citrine line, we follow Dmd localization to the myosepta in real-time using time-lapse microscopy, and find that the accumulation of Dmd protein at the transverse myosepta coincides with the onset of myotome formation, a critical stage in muscle maturation. We observed that Dmd protein localizes specifically to the myosepta prior to dmd mRNA localization. Additionally, we demonstrate that the Dmd-citrine line can be used to assess muscular dystrophy following both genetic and physical disruptions of the muscle.
No takes yet. Share an insight, caveat, or question.
Ruf-Zamojski et al. (2015) studied Muscular dystrophy. Dmd-citrine gene trap line was evaluated on Dmd protein localization and dynamics. A novel zebrafish Dmd-citrine gene trap line revealed that Dmd protein localizes specifically to the myosepta prior to dmd mRNA localization during muscle maturation.
Synapse has enriched 5 closely related papers on similar clinical questions. Consider them for comparative context: