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Grillet, M., Dominguez Gonzalez, B., Sicart, A., Pöttler, M., Cascalho, A., Billion, K., Hernandez Diaz, S., Swerts, J., Naismith, T.V., Gounko, N.V., Verstreken, P., Hanson, P.I., Goodchild, R.E. (2016). Torsins Are Essential Regulators of Cellular Lipid Metabolism.  Dev. Cell 38(3): 235--247.
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Torsins are developmentally essential AAA+ proteins, and mutation of human torsinA causes the neurological disease DYT1 dystonia. They localize in the ER membranes, but their cellular function remains unclear. We now show that dTorsin is required in Drosophila adipose tissue, where it suppresses triglyceride levels, promotes cell growth, and elevates membrane lipid content. We also see that human torsinA at the inner nuclear membrane is associated with membrane expansion and elevated cellular lipid content. Furthermore, the key lipid metabolizing enzyme, lipin, is mislocalized in dTorsin-KO cells, and dTorsin increases levels of the lipin substrate, phosphatidate, and reduces the product, diacylglycerol. Finally, genetic suppression of dLipin rescues dTorsin-KO defects, including adipose cell size, animal growth, and survival. These findings identify that torsins are essential regulators of cellular lipid metabolism and implicate disturbed lipid biology in childhood-onset DYT1 dystonia.

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Personal communication to FlyBase

CG14103 nomenclature.
Goodchild, 2016.10.6, CG14103 nomenclature. [FBrf0233789]


Role for Torsin in Lipid Metabolism.
Teleman, 2016, Dev. Cell 38(3): 223--224 [FBrf0233118]

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    Publication Type
    Dev. Cell
    Developmental Cell
    Publication Year
    1534-5807 1878-1551
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