The effects of ER morphology on synaptic structure and function in Drosophila melanogaster

James B. Summerville, Joseph F. Faust, Ethan Fan, Diana Pendin, Andrea Daga, Joseph Formella, Michael Stern, James A. McNew

Research output: Contribution to journalArticlepeer-review


Hereditary spastic paraplegia (HSP) is a set of genetic diseases caused by mutations in one of 72 genes that results in age-dependent corticospinal axon degeneration accompanied by spasticity and paralysis. Two genes implicated in HSPs encode proteins that regulate endoplasmic reticulum (ER) morphology. Atlastin 1 (ATL1, also known as SPG3A) encodes an ER membrane fusion GTPase and reticulon 2 (RTN2, also known as SPG12) helps shape ER tube formation. Here, we use a new fluorescent ER marker to show that the ER within wild-type Drosophila motor nerve terminals forms a network of tubules that is fragmented and made diffuse upon loss of the atlastin 1 ortholog atl. atl or Rtnl1 loss decreases evoked transmitter release and increases arborization. Similar to other HSP proteins, Atl inhibits bone morphogenetic protein (BMP) signaling, and loss of atl causes age-dependent locomotor deficits in adults. These results demonstrate a crucial role for ER in neuronal function, and identify mechanistic links between ER morphology, neuronal function, BMP signaling and adult behavior.

Original languageEnglish
Pages (from-to)1635-1648
Number of pages14
JournalJournal of Cell Science
Issue number8
Publication statusPublished - Apr 1 2016


  • Atlastin
  • ER
  • Neuron
  • Reticulon

ASJC Scopus subject areas

  • Cell Biology

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