Here, we examined the expression of ceramide metabolism enzymes in the subcutaneous adipose tissue (SAT), epicardial adipose tissue (EAT) and perivascular adipose tissue (PVAT) of 30 patients with coronary artery disease (CAD) and 30 patients with valvular heart disease (VHD) by means of quantitative polymerase chain reaction and fluorescent Western blotting. The EAT of patients with CAD showed higher expression of the genes responsible for ceramide biosynthesis (SPTLC1,SPTLC2,CERS1,5,6,DEGS1, andSMPD1) and utilization (ASAH1,SGMS1). PVAT was characterized by higher mRNA levels ofCERS3,CERS4,DEGS1,SMPD1, and ceramide utilization enzyme (SGMS2). In patients with VHD, there was a highCERS4,DEGS1, andSGMS2expression in the EAT andCERS3andCERS4expression in the PVAT. Among patients with CAD, the expression ofSPTLC1in SAT and EAT,SPTLC2in EAT,CERS2in all studied AT,CERS4andCERS5in EAT,DEGS1in SAT and EAT,ASAH1in all studied AT, andSGMS1in EAT was higher than in those with VHD. Protein levels of ceramide-metabolizing enzymes were consistent with gene expression trends. The obtained results indicate an activation of ceramide synthesis de novo and from sphingomyelin in cardiovascular disease, mainly in EAT, that contributes to the accumulation of ceramides in this location.