In this case report, we present the radiologic and pathologic findings of atypical ductal hyperplasia (ADH) in the malebreast. It is well known that a high-risk lesion such as ADH is a precursor of breast cancer in females. However, the clinical significance of these lesions in the malebreast is still uncertain because malebreasts mainly consist of ducts without lobule formation, unlike the femalebreast. To our knowledge, imaging findings of ADH in the malebreast have not been reported previously, except for a few studies on the pathologic findings of these lesions. Through this paper, we would like to present the possible imaging features of this high-risk lesion in the malebreast and review the related literature.