Presynaptic dysfunction in Drosophila csp mutants

Neuron. 1994 Oct;13(4):899-907. doi: 10.1016/0896-6273(94)90255-0.


Cysteine string proteins are synapse-specific proteins. In Drosophila, csp deletion mutants exhibit temperature-sensitive paralysis and early death. Here, we report that neuromuscular transmission is impaired presynaptically in these csp mutant larvae. At 22 degrees C, evoked transmitter release is depressed relative to wild type and rescued controls, and high frequency stimulation of the nerve leads to sporadic failures. At 30 degrees C, stimulus-evoked responses decline gradually before failing completely. When the temperature is returned to 22 degrees C, evoked responses recover. Spontaneous release events persist at both 22 degrees C and 30 degrees C. Since nerve conduction and postsynaptic sensitivity are unaffected, these data indicate that csp mutations disrupt depolarization-secretion coupling. This disruption explains the cellular basis of the temperature-sensitive paralysis of these organisms.

Publication types

  • Research Support, Non-U.S. Gov't
  • Research Support, U.S. Gov't, Non-P.H.S.
  • Research Support, U.S. Gov't, P.H.S.

MeSH terms

  • Animals
  • Calcium / pharmacology
  • Drosophila / genetics
  • Drosophila / physiology*
  • Evoked Potentials / drug effects
  • Gene Deletion
  • HSP40 Heat-Shock Proteins
  • Larva / physiology
  • Membrane Proteins*
  • Mutation*
  • Nerve Tissue Proteins / genetics*
  • Nerve Tissue Proteins / physiology
  • Neuromuscular Junction / physiology
  • Synapses / physiology*
  • Synaptic Membranes / physiology
  • Synaptic Transmission
  • Temperature


  • HSP40 Heat-Shock Proteins
  • Membrane Proteins
  • Nerve Tissue Proteins
  • cysteine string protein
  • Calcium