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lüll Fatty acid 2-Hydroxylation in mammalian sphingolipid biology Hama HBiochim Biophys Acta 2010[Apr]; 1801 (4): 405-142-Hydroxy fatty acids (hFA) are important components of a subset of mammalian sphingolipids. The presence of hFA in sphingolipids is best described in the nervous system, epidermis, and kidney. However, the literature also indicates that various hFA-sphingolipids are present in additional tissues and cell types, as well as in tumors. Biosynthesis of hFA-sphingolipids requires fatty acid 2-hydroyxlase, and degradation of hFA-sphingolipids depends, at least in part, on lysosomal acid ceramidase and the peroxisomal fatty acid alpha-oxidation pathway. Mutations in the fatty acid 2-hydroxylase gene, FA2H, have been associated with leukodystrophy and spastic paraparesis in humans, underscoring the importance of hFA-sphingolipids in the nervous system. In the epidermis, hFA-ceramides are essential for the permeability barrier function. Physiological function of hFA-sphingolipids in other organs remains largely unknown. Recent evidence indicates that hFA-sphingolipids have specific roles in cell signaling.|Animals[MESH]|Fatty Acids/*metabolism[MESH]|Humans[MESH]|Hydroxylation[MESH]|Mixed Function Oxygenases/*metabolism[MESH]|Sphingolipids/*metabolism[MESH] |