Distinct roles for telethonin N-versus C-terminus in sarcomere assembly and maintenance

Dev Dyn. 2010 Apr;239(4):1124-35. doi: 10.1002/dvdy.22263.

Abstract

The N-terminus of telethonin forms a unique structure linking two titin N-termini at the Z-disc. While a specific role for the C-terminus has not been established, several studies indicate it may have a regulatory function. Using a morpholino approach in Xenopus, we show that telethonin knockdown leads to embryonic paralysis, myocyte defects, and sarcomeric disruption. These myopathic defects can be rescued by expressing full-length telethonin mRNA in morpholino background, indicating that telethonin is required for myofibrillogenesis. However, a construct missing C-terminal residues is incapable of rescuing motility or sarcomere assembly in cultured myocytes. We, therefore, tested two additional constructs: one where four C-terminal phosphorylatable residues were mutated to alanines and another where terminal residues were randomly replaced. Data from these experiments support that the telethonin C-terminus is required for assembly, but in a context-dependent manner, indicating that factors and forces present in vivo can compensate for C-terminal truncation or mutation.

MeSH terms

  • Amino Acid Sequence
  • Animals
  • Animals, Genetically Modified
  • Embryo, Nonmammalian
  • Gene Expression Regulation, Developmental / drug effects
  • Gene Expression Regulation, Developmental / genetics
  • Gene Expression Regulation, Developmental / physiology
  • Models, Molecular
  • Muscle Development / drug effects
  • Muscle Development / genetics*
  • Muscle Development / physiology
  • Muscle Fibers, Skeletal / metabolism
  • Muscle Fibers, Skeletal / physiology
  • Muscle Proteins / chemistry*
  • Muscle Proteins / genetics
  • Muscle Proteins / physiology*
  • Muscle, Striated / growth & development
  • Muscle, Striated / metabolism
  • Muscle, Striated / physiology
  • Oligoribonucleotides, Antisense / pharmacology
  • Protein Structure, Tertiary / genetics
  • Protein Structure, Tertiary / physiology
  • Sarcomeres / drug effects
  • Sarcomeres / genetics
  • Sarcomeres / metabolism*
  • Sarcomeres / physiology
  • Sequence Homology
  • Xenopus

Substances

  • Muscle Proteins
  • Oligoribonucleotides, Antisense