Summerville, J.B., Faust, J.F., Fan, E., Pendin, D., Daga, A., Formella, J., Stern, M., McNew, J.A. (2016). The effects of ER morphology on synaptic structure and function in Drosophila melanogaster. J. Cell Sci. 129(8): 1635--1648.
FlyBase ID
FBrf0232027
Publication Type
Research paper
Abstract
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-typeDrosophilamotor nerve terminals forms a network of tubules that is fragmented and made diffuse upon loss of the atlastin 1 orthologatlatlorRtnl1loss decreases evoked transmitter release and increases arborization. Similar to other HSP proteins, Atl inhibits bone morphogenetic protein (BMP) signaling, and loss ofatlcauses 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.
