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The atrial structure or substrate of patients with atrial arrhythmias can present very differently, and also, the “phenotype” of the arrhythmia cannot explain these differences. Idiopathic isolated atrial cardiomyopathy is a novel subtype of cardiomyopathy characterized possibly by atrial fibrosis that does not involve the ventricular myocardium and is associated with significant atrial tachyarrhythmia and no systemic, inflammatory, or infiltrative disease. We report a 57-year-old female doctor who presented with recurrent palpitations of 2-week duration and was shown to have normal left ventricular size, function, longitudinal strain, and filling pressures as judged by E/e' ratio and mitral early diastolic Doppler tissue velocities. The study of atria revealed markedly enlarged left atrium (LA), significantly reduced left atrial reservoir strain, and increased LA stiffness index. Frequent atrial ectopy may be manifestation of a structural atrial disease which could be termed arrhythmogenic isolated LA cardiomyopathy.