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PMID:19341724

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Citation

Lee, M, Paik, SK, Lee, MJ, Kim, YJ, Kim, S, Nahm, M, Oh, SJ, Kim, HM, Yim, J, Lee, CJ, Bae, YC and Lee, S (2009) Drosophila Atlastin regulates the stability of muscle microtubules and is required for synapse development. Dev. Biol. 330:250-62

Abstract

Hereditary spastic paraplegia (HSP) is an inherited neurological disorder characterized by progressive spasticity and weakness of the lower extremities. The most common early-onset form of HSP is caused by mutations in the human gene that encodes the dynamin-family GTPase Atlastin-1 (Atl-1). Recently, loss of the Drosophila ortholog of Atl-1 (Atl) has been found to induce locomotor impairments from the earliest adult stages, suggesting the developmental role of atlastin-subfamily GTPases. Here, we provide evidence that Atl is required for normal growth of muscles and synapses at the neuromuscular junction (NMJ). Atl protein is highly expressed in larval body-wall muscles. Loss-of-function mutations in the atl gene reduce the size of muscles and increase the number of synaptic boutons. Rescue of these defects is accomplished by muscular, but not neuronal expression of Atl. Loss of Atl also disrupts ER and Golgi morphogenesis in muscles and reduces the synaptic levels of the scaffold proteins Dlg and alpha-spectrin. We also provide evidence that Atl functions with the microtubule-severing protein Spastin to disassemble microtubules in muscles. Finally, we demonstrate that the microtubule-destabilizing drug vinblastine alleviates synapse and muscle defects in atl mutants. Together, our results suggest that Atl controls synapse development and ER and Golgi morphogenesis by regulating microtubule stability.

Links

PubMed Online version:10.1016/j.ydbio.2009.03.019

Keywords

Animals; Drosophila Proteins/genetics; Drosophila Proteins/physiology; Drosophila melanogaster/embryology; Drosophila melanogaster/genetics; Microtubules; Morphogenesis; Muscles/embryology; Mutation; Synapses

Significance

Annotations

Gene product Qualifier GO Term Evidence Code with/from Aspect Extension Notes Status

DROME:SPAST

enables

GO:0005515: protein binding

ECO:0000353: physical interaction evidence used in manual assertion

UniProtKB:Q9VC57

F

Seeded From UniProt

complete

DROME:ATLAS

enables

GO:0005515: protein binding

ECO:0000353: physical interaction evidence used in manual assertion

UniProtKB:Q8I0P1

F

Seeded From UniProt

complete

DROME:ATLAS

located_in

GO:0005794: Golgi apparatus

ECO:0000314: direct assay evidence used in manual assertion

C

Seeded From UniProt

complete

DROME:ATLAS

involved_in

GO:0051124: synaptic assembly at neuromuscular junction

ECO:0000315: mutant phenotype evidence used in manual assertion

P

Seeded From UniProt

complete

DROME:ATLAS

located_in

GO:0005783: endoplasmic reticulum

ECO:0000314: direct assay evidence used in manual assertion

C

Seeded From UniProt

complete

DROME:ATLAS

involved_in

GO:0008582: regulation of synaptic assembly at neuromuscular junction

ECO:0000315: mutant phenotype evidence used in manual assertion

P

Seeded From UniProt

complete

DROME:ATLAS

involved_in

GO:0008582: regulation of synaptic growth at neuromuscular junction

ECO:0000315: mutant phenotype evidence used in manual assertion

P

Seeded From UniProt

complete

DROME:ATLAS

involved_in

GO:0031114: regulation of microtubule depolymerization

ECO:0000315: mutant phenotype evidence used in manual assertion

P

Seeded From UniProt

complete

DROME:ATLAS

involved_in

GO:0007528: neuromuscular junction development

ECO:0000315: mutant phenotype evidence used in manual assertion

P

Seeded From UniProt

complete

DROME:ATLAS

part_of

GO:0005783: endoplasmic reticulum

ECO:0000314: direct assay evidence used in manual assertion

C

Seeded From UniProt

complete

DROME:ATLAS

part_of

GO:0005794: Golgi apparatus

ECO:0000314: direct assay evidence used in manual assertion

C

Seeded From UniProt

complete

DROME:ATLAS

colocalizes_with

GO:0005874: microtubule

ECO:0000314: direct assay evidence used in manual assertion

C

Seeded From UniProt

complete

DROME:ATLAS

involved_in

GO:0007019: microtubule depolymerization

ECO:0000315: mutant phenotype evidence used in manual assertion

P

Seeded From UniProt

complete

DROME:ATLAS

involved_in

GO:0007029: endoplasmic reticulum organization

ECO:0000315: mutant phenotype evidence used in manual assertion

P

Seeded From UniProt

complete

DROME:ATLAS

involved_in

GO:0007030: Golgi organization

ECO:0000315: mutant phenotype evidence used in manual assertion

P

Seeded From UniProt

complete

DROME:ATLAS

involved_in

GO:0007517: muscle organ development

ECO:0000315: mutant phenotype evidence used in manual assertion

P

Seeded From UniProt

complete

DROME:ATLAS

involved_in

GO:0051124: synaptic growth at neuromuscular junction

ECO:0000315: mutant phenotype evidence used in manual assertion

P

Seeded From UniProt

complete


See also

References

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