ArticleDevelopment (Cambridge, England)2013
Genetic elevation of sphingosine 1-phosphate suppresses dystrophic muscle phenotypes in Drosophila.
Article in Development (Cambridge, England), 2013. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 28 papers.
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The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.
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Who cites it
28 citing papers in PubMed, 37 citations in OpenAlex.
- Credit to the Fruit Fly: How the Tiny Insect Lights Up Our Understanding of Human Disease.Insects · 2026Review
- TET exhibits enzymatic-independent and-dependent functions during Drosophila flight muscle development and aging.Skeletal muscle · 2025Article
- Synaptic defects in a drosophila model of muscular dystrophy.Frontiers in cellular neuroscience · 2024Article
- Very-long-chain fatty acids induce glial-derived sphingosine-1-phosphate synthesis, secretion, and neuroinflammation.Cell metabolism · 2023Article
- Gpr18 agonist dampens inflammation, enhances myogenesis, and restores muscle function in models of Duchenne muscular dystrophy.Frontiers in cell and developmental biology · 2023Article
- Sphingosine Phosphate Lyase Is Upregulated in Duchenne Muscular Dystrophy, and Its Inhibition Early in Life Attenuates Inflammation and Dystrophy in Mdx Mice.International journal of molecular sciences · 2022Article
- Diverse roles of phosphatidate phosphatases in insect development and metabolism.Insect biochemistry and molecular biology · 2021Review
- In the Model HostInternational journal of molecular sciences · 2020Article
- Visualizing Synaptic Degeneration in Adult Drosophila in Association with Neurodegeneration.Journal of visualized experiments : JoVE · 2020Article
- Duchenne muscular dystrophy animal models for high-throughput drug discovery and precision medicine.Expert opinion on drug discovery · 2020Review
- Sphingolipid Metabolism and Signaling in Skeletal Muscle: From Physiology to Physiopathology.Frontiers in endocrinology · 2020Review
- Adult Drosophila muscle morphometry through microCT reveals dynamics during ageing.Open biology · 2019Article
- Lipid metabolic perturbation is an early-onset phenotype in adultMolecular biology of the cell · 2017Article
- Overexpression of miRNA-9 Generates Muscle Hypercontraction Through Translational Repression of Troponin-T inG3 (Bethesda, Md.) · 2017Article
- Severe murine limb-girdle muscular dystrophy type 2C pathology is diminished by FTY720 treatment.Muscle & nerve · 2017Article
- miR-127 enhances myogenic cell differentiation by targeting S1PR3.Cell death & disease · 2017Article
- Pathway to a phenocopy: Heat stress effects in early embryogenesis.Developmental dynamics : an official publication of the American Association of Anatomists · 2016Article
- Model organisms in the fight against muscular dystrophy: lessons from drosophila and Zebrafish.Molecules (Basel, Switzerland) · 2015Review
- Silencing of drpr leads to muscle and brain degeneration in adult Drosophila.The American journal of pathology · 2014Article
- Recent advances using zebrafish animal models for muscle disease drug discovery.Expert opinion on drug discovery · 2014Review
Corrections and comments
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Authors and funding
5 authors at 1 institution in 1 country.
Funding
Abstract
Duchenne muscular dystrophy is a lethal genetic disease characterized by the loss of muscle integrity and function over time. Using Drosophila, we show that dystrophic muscle phenotypes can be significantly suppressed by a reduction of wunen, a homolog of lipid phosphate phosphatase 3, which in higher animals can dephosphorylate a range of phospholipids. Our suppression analyses include assessing the localization of Projectin protein, a titin homolog, in sarcomeres as well as muscle morphology and functional movement assays. We hypothesize that wunen-based suppression is through the elevation of the bioactive lipid Sphingosine 1-phosphate (S1P), which promotes cell proliferation and differentiation in many tissues, including muscle. We confirm the role of S1P in suppression by genetically altering S1P levels via reduction of S1P lyase (Sply) and by upregulating the serine palmitoyl-CoA transferase catalytic subunit gene lace, the first gene in the de novo sphingolipid biosynthetic pathway and find that these manipulations also reduce muscle degeneration. Furthermore, we show that reduction of spinster (which encodes a major facilitator family transporter, homologs of which in higher animals have been shown to transport S1P) can also suppress dystrophic muscle degeneration. Finally, administration to adult flies of pharmacological agents reported to elevate S1P signaling significantly suppresses dystrophic muscle phenotypes. Our data suggest that localized intracellular S1P elevation promotes the suppression of muscle wasting in flies.
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Registered trials
Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.