Intraductal papillomas may rarely contain areas of atypical epithelial hyperplasia and ductal carcinoma in situ. The information about clinical outcome of intraductal papillomas with features of atypical ductal hyperplasia-ductal carcinoma in situ is inadequate. In this study, 59 intraductal papilloma cases belonging to 55 patients were reviewed retrospectively according to archives records and slides. Atypical epithelial hyperplasia was observed in 12 of the fifty-nine intraductal papillomas (20 %). While two of the intraductal papilloma with atypical epithelial hyperplasia developed carcinoma on the same side (16 %), papilloma without atypia developing carcinoma was only 2%. In the patients who had followed-up, recurrence was 13 %. It was thought that intraductal papilloma with atypical epithelial hyperplasia related to recurrence and carcinomatous transformation.