Sphingolipids Regulate Neuromuscular Synapse Structure and function in Drosophila

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Sphingolipids Regulate Neuromuscular Synapse Structure and function in Drosophila. / West, Ryan John Hatcher; Briggs, Laura; Fjeldstad, Maria Perona; Ribchester, Richard R.; Sweeney, Sean.

In: Journal of comparative neurology, 01.09.2018.

Research output: Contribution to journalArticle

Harvard

West, RJH, Briggs, L, Fjeldstad, MP, Ribchester, RR & Sweeney, S 2018, 'Sphingolipids Regulate Neuromuscular Synapse Structure and function in Drosophila', Journal of comparative neurology. https://doi.org/10.1002/cne.24466

APA

West, R. J. H., Briggs, L., Fjeldstad, M. P., Ribchester, R. R., & Sweeney, S. (2018). Sphingolipids Regulate Neuromuscular Synapse Structure and function in Drosophila. Journal of comparative neurology. https://doi.org/10.1002/cne.24466

Vancouver

West RJH, Briggs L, Fjeldstad MP, Ribchester RR, Sweeney S. Sphingolipids Regulate Neuromuscular Synapse Structure and function in Drosophila. Journal of comparative neurology. 2018 Sep 1. https://doi.org/10.1002/cne.24466

Author

West, Ryan John Hatcher ; Briggs, Laura ; Fjeldstad, Maria Perona ; Ribchester, Richard R. ; Sweeney, Sean. / Sphingolipids Regulate Neuromuscular Synapse Structure and function in Drosophila. In: Journal of comparative neurology. 2018.

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@article{12b880174ace4f41b1945548c90b7fa4,
title = "Sphingolipids Regulate Neuromuscular Synapse Structure and function in Drosophila",
abstract = "Sphingolipids are found in abundance at synapses and have been implicated inregulation of synapse structure, function and degeneration. Their precise role inthese processes, however, remains obscure. Serine Palmitoyl-transferase (SPT)is the first enzymatic step for synthesis of sphingolipids. Analysis of theDrosophila larval neuromuscular junction revealed mutations in the SPT enzymesubunit, lace/SPTLC2 resulted in deficits in synaptic structure and function.Although neuromuscular junction (NMJ) length is normal in lace mutants, thenumber of boutons per NMJ is reduced to ~50{\%} of the wild type number.Synaptic boutons in lace mutants are much larger but show little perturbation tothe general ultrastructure. Electrophysiological analysis of lace mutant synapsesrevealed strong synaptic transmission coupled with predominance of depressionover facilitation. The structural and functional phenotypes of lace mirroredaspects of Basigin (Bsg), a small Ig-domain adhesion molecule also known toregulate synaptic structure and function. Mutant combinations of lace and Bsggenerated large synaptic boutons, while lace mutants showed abnormalaccumulation of Bsg at synapses, suggesting that Bsg requires sphingolipid toregulate structure of the synapse. In support of this, we found Bsg to be enriched in lipid rafts. Our data points to a role for sphingolipids in the regulation and fine-tuning of synaptic structure and function while sphingolipid regulation of synaptic structure may be mediated via the activity of Bsg.",
author = "West, {Ryan John Hatcher} and Laura Briggs and Fjeldstad, {Maria Perona} and Ribchester, {Richard R.} and Sean Sweeney",
note = "This is an author-produced version of the published paper. Uploaded in accordance with the publisher’s self-archiving policy. Further copying may not be permitted; contact the publisher for details",
year = "2018",
month = "9",
day = "1",
doi = "10.1002/cne.24466",
language = "English",
journal = "Journal of comparative neurology",
issn = "0021-9967",
publisher = "Wiley-Liss Inc.",

}

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TY - JOUR

T1 - Sphingolipids Regulate Neuromuscular Synapse Structure and function in Drosophila

AU - West, Ryan John Hatcher

AU - Briggs, Laura

AU - Fjeldstad, Maria Perona

AU - Ribchester, Richard R.

AU - Sweeney, Sean

N1 - This is an author-produced version of the published paper. Uploaded in accordance with the publisher’s self-archiving policy. Further copying may not be permitted; contact the publisher for details

PY - 2018/9/1

Y1 - 2018/9/1

N2 - Sphingolipids are found in abundance at synapses and have been implicated inregulation of synapse structure, function and degeneration. Their precise role inthese processes, however, remains obscure. Serine Palmitoyl-transferase (SPT)is the first enzymatic step for synthesis of sphingolipids. Analysis of theDrosophila larval neuromuscular junction revealed mutations in the SPT enzymesubunit, lace/SPTLC2 resulted in deficits in synaptic structure and function.Although neuromuscular junction (NMJ) length is normal in lace mutants, thenumber of boutons per NMJ is reduced to ~50% of the wild type number.Synaptic boutons in lace mutants are much larger but show little perturbation tothe general ultrastructure. Electrophysiological analysis of lace mutant synapsesrevealed strong synaptic transmission coupled with predominance of depressionover facilitation. The structural and functional phenotypes of lace mirroredaspects of Basigin (Bsg), a small Ig-domain adhesion molecule also known toregulate synaptic structure and function. Mutant combinations of lace and Bsggenerated large synaptic boutons, while lace mutants showed abnormalaccumulation of Bsg at synapses, suggesting that Bsg requires sphingolipid toregulate structure of the synapse. In support of this, we found Bsg to be enriched in lipid rafts. Our data points to a role for sphingolipids in the regulation and fine-tuning of synaptic structure and function while sphingolipid regulation of synaptic structure may be mediated via the activity of Bsg.

AB - Sphingolipids are found in abundance at synapses and have been implicated inregulation of synapse structure, function and degeneration. Their precise role inthese processes, however, remains obscure. Serine Palmitoyl-transferase (SPT)is the first enzymatic step for synthesis of sphingolipids. Analysis of theDrosophila larval neuromuscular junction revealed mutations in the SPT enzymesubunit, lace/SPTLC2 resulted in deficits in synaptic structure and function.Although neuromuscular junction (NMJ) length is normal in lace mutants, thenumber of boutons per NMJ is reduced to ~50% of the wild type number.Synaptic boutons in lace mutants are much larger but show little perturbation tothe general ultrastructure. Electrophysiological analysis of lace mutant synapsesrevealed strong synaptic transmission coupled with predominance of depressionover facilitation. The structural and functional phenotypes of lace mirroredaspects of Basigin (Bsg), a small Ig-domain adhesion molecule also known toregulate synaptic structure and function. Mutant combinations of lace and Bsggenerated large synaptic boutons, while lace mutants showed abnormalaccumulation of Bsg at synapses, suggesting that Bsg requires sphingolipid toregulate structure of the synapse. In support of this, we found Bsg to be enriched in lipid rafts. Our data points to a role for sphingolipids in the regulation and fine-tuning of synaptic structure and function while sphingolipid regulation of synaptic structure may be mediated via the activity of Bsg.

U2 - 10.1002/cne.24466

DO - 10.1002/cne.24466

M3 - Article

JO - Journal of comparative neurology

JF - Journal of comparative neurology

SN - 0021-9967

ER -