Two cases of myopathy associated with ezetimibe are reported. In the first case, a woman on ezetimibe monotherapy presented with muscle pain and an elevated concentration of creatine kinase (CK) on two occasions, with ezetimibe 10 mg and with ezetimibe 5 mg after a washout period. The recurrence of muscle pain after washout and the CK increase both supported the hypothesis that ezetimibe alone can be linked to myalgia. In the second case, a man had been treated with atorvastatin, and ezetimibe 10 mg was added to improve his lipid profile. Two months later, the patient complained of muscle pain and a CK increase was noted. The appearance of symptoms when adding ezetimibe to atorvastatin supports a potential pharmacokinetic and/or a pharmacodynamic interaction between these two drugs. These cases suggest that ezetimibe monotherapy as well as ezetimibe associated with the use of a statin may induce myalgia. The mechanism by which ezetimibe could cause muscle pain is not known.