Drosophila Atlastin regulates the stability of muscle microtubules and is required for synapse development.

  title={Drosophila Atlastin regulates the stability of muscle microtubules and is required for synapse development.},
  author={Mihye Lee and Sang Kyoo Paik and Min-Jung Lee and Yoon Jung Kim and Sungdae Kim and Minyeop Nahm and Soo Jin Oh and Hyun-Man Kim and Jeongbin Yim and C. Justin Lee and Yong Chul Bae and Seungbok Lee},
  journal={Developmental biology},
  volume={330 2},

Drosophila Atlastin in motor neurons is required for locomotion and presynaptic function

It is suggested that trafficking defects produced by Atlastin dysfunction in motor neurons result in redistribution of presynaptic components and aberrant mobilization of synaptic vesicles, stressing the importance of ER-shaping proteins and the susceptibility of motor neurons to their mutations or depletion.

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

A new fluorescent ER marker is used 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.

Drosophila Atlastin regulates synaptic vesicle mobilization independent of Bone Morphogenetic Protein signaling

A mechanism by which the loss of an ER structuring protein in the motor neuron could, through its role in regulating SV and endosomal trafficking, explain defects in SV accumulation and synaptic dysfunction is suggested.

Swiss Cheese, Drosophila Ortholog of Hereditary Spastic Paraplegia Gene NTE, Maintains Neuromuscular Junction Development and Microtubule Network

It is established that mutations in the sws gene alter NMJ morphology, the distribution of synaptic markers, microtubule (MT) network, and synaptic microtubules organization, and it is shown that sws is widely expressed in the larval nervous system, especially in glial cells.

Pharmacologic rescue of axon growth defects in a human iPSC model of hereditary spastic paraplegia SPG3A.

The prominent alterations in axon growth in SPG3A neurons may represent a particularly attractive target for suppression in screens for novel pharmacologic agents, emphasizing the importance of tubular ER interactions with the microtubule cytoskeleton in hereditary spastic paraplegia pathogenesis.

Beneficial effects of rapamycin in a Drosophila model for hereditary spastic paraplegia

It is shown that Drosophila lacking atl, a gene associated with hereditary spastic paraplegia, exhibit muscle degeneration and other phenotypes that are each rescued by administering the Tor inhibitor rapamycin, indicating that atl loss triggers Muscle degeneration both cell autonomously and nonautonomously.

autosomal-recessive form of HSP ( SPG 39 )

It is shown that mutations in sws (sws and sws76−1 and SWS knockdown alter neuromuscular junction’s morphology and synaptic microtubules organization, which leads to age-dependent neurodegeneration, structure alteration of glia cells, and reduced insect life span.

The BMP signaling pathway at the Drosophila neuromuscular junction and its links to neurodegenerative diseases




Atlastin GTPases are required for Golgi apparatus and ER morphogenesis.

The atlastin family of GTPases functions prominently in both ER and Golgi morphogenesis, but they do not appear to be required generally for anterograde ER-to-Golgi trafficking.

The microtubule-severing protein Spastin is essential for axon outgrowth in the zebrafish embryo.

A critical requirement for spastin to promote axonal outgrowth during embryonic development is revealed, and the zebrafish embryo is validated as a novel model system to dissect the pathogenetic mechanisms underlying HSP.

SPG3A protein atlastin-1 is enriched in growth cones and promotes axon elongation during neuronal development.

It is demonstrated that several missense SPG3A mutant atlastin-1 proteins have impaired GTPase activity and thus may act in a dominant-negative, loss-of-function manner by forming mixed oligomers with wild-type atlastIn-1, prefiguring a functional role for atlast in-1 in axonal development.

The Drosophila β-Amyloid Precursor Protein Homolog Promotes Synapse Differentiation at the Neuromuscular Junction

It is shown that APPL was transported to motor axons and that its overexpression caused a dramatic increase in synaptic bouton number and changes in synapse structure, and a model by which APPL, in conjunction with activity-dependent mechanisms, regulates synaptic structure and number is proposed.

Interaction of two hereditary spastic paraplegia gene products, spastin and atlastin, suggests a common pathway for axonal maintenance.

A physical interaction between spastin and atlastin is shown, indicating that at least a subset of HSP genes may define a cellular biological pathway that is important in axonal maintenance.

Loss of spastic paraplegia gene atlastin induces age-dependent death of dopaminergic neurons in Drosophila

Spastin and atlastin, two proteins mutated in autosomal-dominant hereditary spastic paraplegia, are binding partners.

It is shown that the spastin domain required for binding to atlastin lies within the N-terminal 80 residues of the protein, a region that is only present in the predominantly cytoplasmic, full-length spastIn isoform.

Cellular Localization, Oligomerization, and Membrane Association of the Hereditary Spastic Paraplegia 3A (SPG3A) Protein Atlastin*

Findings indicate that the SPG3A protein atlastin-1 is a multimeric integral membrane GTPase that may be involved in Golgi membrane dynamics or vesicle trafficking.