Reference - Detail
| RRC ID | 45657 |
|---|---|
| Author | Xie M, Roy R. |
| Title | The Causative Gene in Chanarian Dorfman Syndrome Regulates Lipid Droplet Homeostasis in C. elegans. |
| Journal | PLoS Genet |
| Abstract |
AMP-activated kinase (AMPK) is a key regulator of many cellular mechanisms required for adjustment to various stresses induced by the changing environment. In C. elegans dauer larvae AMPK-null mutants expire prematurely due to hyperactive Adipose Triglyceride Lipase (ATGL-1) followed by rapid depletion of triglyceride stores. We found that the compromise of one of the three C. elegans orthologues of human cgi-58 significantly improves the survival of AMPK-deficient dauers. We also provide evidence that C. elegans CGI-58 acts as a co-activator of ATGL-1, while it also functions cooperatively to maintain regular lipid droplet structure. Surprisingly, we show that it also acts independently of ATGL-1 to restrict lipid droplet coalescence by altering the surface abundance and composition of long chain (C20) polyunsaturated fatty acids (PUFAs). Our data reveal a novel structural role of CGI-58 in maintaining lipid droplet homeostasis through its effects on droplet composition, morphology and lipid hydrolysis; a conserved function that may account for some of the ATGL-1-independent features unique to Chanarin-Dorfman Syndrome. |
| Volume | 11(6) |
| Pages | e1005284 |
| Published | 2015-6-1 |
| DOI | 10.1371/journal.pgen.1005284 |
| PII | PGENETICS-D-14-03370 |
| PMID | 26083785 |
| PMC | PMC4470697 |
| MeSH | 1-Acylglycerol-3-Phosphate O-Acyltransferase / genetics* 1-Acylglycerol-3-Phosphate O-Acyltransferase / metabolism Amino Acid Sequence Animals Caenorhabditis elegans / genetics* Caenorhabditis elegans / metabolism Caenorhabditis elegans Proteins / genetics Caenorhabditis elegans Proteins / metabolism Fatty Acids, Unsaturated / metabolism Homeostasis Ichthyosiform Erythroderma, Congenital / genetics* Lipase / genetics Lipase / metabolism Lipid Droplets / metabolism* Lipid Metabolism, Inborn Errors / genetics* Molecular Sequence Data Muscular Diseases / genetics* |
| IF | 5.175 |
| Times Cited | 12 |
| WOS Category | GENETICS & HEREDITY |
| Altmetric score |
オルトメトリクス指標項目
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| The most frequently cited source | X(Twitter) |
| Total number of mentions | 3 |
| Altmetric score changes over past 6months | 0.0 |
| Resource | |
| C.elegans | tm1944 |