RT Journal Article
SR Electronic
T1 Modeling of axonal endoplasmic reticulum network by spastic paraplegia proteins
JF bioRxiv
FD Cold Spring Harbor Laboratory
SP 069005
DO 10.1101/069005
A1 Belgin Yalçın
A1 Lu Zhao
A1 Martin Stofanko
A1 Niamh C. O’Sullivan
A1 Zi Han Kang
A1 Annika Roost
A1 Matthew R. Thomas
A1 Sophie Zaessinger
A1 Olivier Blard
A1 Alex L. Patto
A1 Valentina Baena
A1 Mark Terasaki
A1 Cahir J. O’Kane
YR 2016
UL http://biorxiv.org/content/early/2016/11/17/069005.abstract
AB Axons contain an endoplasmic reticulum (ER) network that is largely smooth and tubular, thought to be continuous with ER throughout the neuron, and distinct in form and function from rough ER; the mechanisms that form this continuous network in axons are not well understood. Mutations affecting proteins of the reticulon or REEP families, which contain intramembrane hairpin domains that can model ER membranes, cause an axon degenerative disease, hereditary spastic paraplegia (HSP). Here, we show that these proteins are required for modeling the axonal ER network in Drosophila. Loss of reticulon or REEP proteins can lead to expansion of ER sheets, and to partial loss of ER from distal motor axons. Ultrastructural analysis reveals an extensive ER network in every axon of peripheral nerves, which is reduced in larvae that lack reticulon and REEP proteins, with defects including larger and fewer tubules, and occasional gaps in the ER network, consistent with loss of membrane curvature. Therefore HSP hairpin-containing proteins are required for shaping and continuity of the axonal ER network, suggesting an important role for ER modeling in axon maintenance and function.