BackgroundAmiodarone is one of the most commonly used and effective antiarrhythmic drugs to treat ventricular and supraventricular arrhythmias. Besides its advantages, this drug has side effects like liver, digestive, pulmonary, thyroid, neural, skin, optical, hematologic, psychiatric, and cardiac complications. Blue-gray cutaneous discoloration, also known as blue man syndrome, is an undesirable and unusual side-effect of chronic amiodarone therapy in less than 3% of patients.Case presentationThis report presents a 51-year-old Caucasian man treated for the past 3 years with amiodarone and implantable cardioverter defibrillators due to his ventricular arrhythmia and cardiomyopathy, without any follow-up visit to his doctor. He was referred to the medical center for blue-gray discoloration on his nose and cheeks, which had started to appear in the last 3 weeks.ConclusionConsidering the findings obtained in this report and the numerous side effects of amiodarone, the blue-man syndrome is a rare yet important finding of this drug which may influence the patient’s daily life. It is recommended that all patients under treatment with this drug be notified about its side effects and visit their doctors regularly. Regarding the high therapeutic value of this drug, the lack of any association between blue man syndrome and other complications, and the related aesthetic problems, the role of the caregiver becomes much more critical in the prescription of amiodarone.