Lipid dysregulation is suggested to participate in the pathogenesis of Alzheimer's disease (AD). However, alterations in individual lipid species in the cerebrospinal fluid (CSF) have not been examined. CSF samples were obtained from 57 AD patients and 68 control, cognitively normal individuals. b-amyloid 1–42 (Ab1–42), phosphorylated tau (P-tau) and total tau (T-tau) proteins were determined by Enzyme-Linked Immunoassay (ELISA) method. Lipid identification and quantification were performed by multi-dimensional mass spectrometry-based Shotgun Lipidomic (MDMS-SL) technique. Levels of long chain sphingomyelin, sulfatide and alkyl phosphatidyl choline were significantly lower for AD than in case of controls, while the level of short chain ceramid, phosphatidyl ethanolamine and phosphatidyl serine were significantly increased in AD. Sphingomyelin and alkyl phosphatidyl choline negatively, while phosphatidyl etanolamine positively correlated with the P-tau level of CFS. These new AD related lipidomic results provide a better understanding of lipid perturbations in AD pathogenesis and may be utilized in the searching for CSF biomarkers. Acknowledgement: This work was supported by the Hungarian Research and Technology Innovation Fund through the Hungarian Brain Research Program (KTIA_13_NAP-A-II/16).