moc-6 /MOCS2A is necessary for molybdenum cofactor synthesis in C. elegans .
| Autor: | Snoozy J; Pediatrics and Rare Diseases Group, Sanford Research, Sioux Falls, SD 57104, USA., Breen PC; Department of Molecular Biology, Massachusetts General Hospital, Boston, MA 02114, USA., Ruvkun G; Department of Molecular Biology, Massachusetts General Hospital, Boston, MA 02114, USA., Warnhoff K; Pediatrics and Rare Diseases Group, Sanford Research, Sioux Falls, SD 57104, USA.; Department of Pediatrics, Sanford School of Medicine, University of South Dakota, Sioux Falls, SD 57105 USA. |
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| Jazyk: | angličtina |
| Zdroj: | MicroPublication biology [MicroPubl Biol] 2022 Feb 22; Vol. 2022. Date of Electronic Publication: 2022 Feb 22 (Print Publication: 2022). |
| DOI: | 10.17912/micropub.biology.000531 |
| Abstrakt: | Molybdenum cofactor (Moco) is an essential prosthetic group that mediates the activity of 4 animal oxidases and is required for viability. Humans with mutations in the genes encoding Moco-biosynthetic enzymes suffer from Moco deficiency, a neonatal lethal inborn error of metabolism. Caenorhabditis elegans has recently emerged as a useful and tractable genetic discovery engine for Moco biology. Here, we identify and characterize K10D2.7/ moc-6 , the C. elegans ortholog of human MOCS2A, a sulfur-carrier protein essential for Moco synthesis. Using CRISPR/Cas9 gene editing, we generate 3 null mutations in K10D2.7/ moc-6 and with these alleles genetically demonstrate that K10D2.7/ moc-6 is necessary for endogenous Moco synthesis in C. elegans. (Copyright: © 2022 by the authors.) |
| Databáze: | MEDLINE |
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