Sphingolipids are involved in a wide range of physiological and pathological processes none only s signaling molecules but also as key structural components regulating the lateral organization of cellular membranes. In this book, exciting new approaches that open a window to further characterize sphingolipid-enriched domains in cell membranes during both physiological and pathological processes are reviewed. Furthermore, sphingolipids (SLs) are especially important in the central nervous system (CNS) where they are a necessary structural component of membranes of brain cells or signaling molecules. Ceramides are the core constituent of most sphingolipids. The authors review the possible sources of ceramides in the central nervous system (CNS) and present information about recent preclinical clinical trials of therapies targeting the ceramide pathway in the brain. Data about significant alteration in levels of ceramides in brain cells during the development of different neurodegenerative disease are also discussed, such as in with Alzheimer’s disease, Parkinson’s disease multiple sclerosis, cerebral ischemia, Gaucher’s and Farber’s disease.