Ductal carcinoma in situ (DCIS) of the male breast is rare. Even more rare is the finding of DCIS in association with gynecomastia. After an extensive literature search, only two cases have been reported in the literature, both in adults. Here we present the case of a 16-year-old adolescent boy who presented with pubertal gynecomastia that was treated with bilateral subcutaneous mastectomies. A DCIS focus was found in the right breast specimen, and the patient underwent bilateral completion total mastectomies uneventfully. Despite its rarity, surgeons should be aware of the possibility of the breast of adolescents with gynecomastia harboring a neoplastic focus.