Abstract
The ceramide synthase (CerS) enzymes catalyze the formation of (dihydro) ceramide, and thereby provide critical complexity to all sphingolipids (SLs) with respect to their acyl chain length. This review summarizes the progress in the field of CerS from the time of their discovery more than a decade ago as Longevity assurance (Lass) genes in yeast, until the recent development of CerS-deficient mouse models. Human hereditary CerS disorders are yet to be discovered. However, the recent findings in CerS mutant animals highlight the important physiological role of these enzymes. The fundamental findings with respect to CerS structure, function, localization, and regulation are discussed, as well as CerS roles in maintaining longevity in vivo.
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- 1.
CerS3 is unique in this respect, as it was found to be important for the last stages of proper skin development, and thus, these mice do not survive after birth (see above).
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Note added in proof:
A recent paper by Zigdon H. et al., (2013) doi: 10.1074/jbc.M112.402719 shows that CerS2 deficiency lead to liver mitochondrial dysfunction, which results in chronic oxidative stress.
Acknowledgments We would like to thank Drs. Anthony H. Futerman, Elad Lavee Laviad, Shifra Ben-Dor, Steffen Jung, and Alexandra Mahler, as well as Oshrit Ben-David and Ruth Seiden, for their constructive comments.
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Park, JW., Pewzner-Jung, Y. (2013). Ceramide Synthases: Reexamining Longevity. In: Gulbins, E., Petrache, I. (eds) Sphingolipids: Basic Science and Drug Development. Handbook of Experimental Pharmacology, vol 215. Springer, Vienna. https://doi.org/10.1007/978-3-7091-1368-4_5
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