We present two cases of cotyledonoid dissecting leiomyoma of the uterus with intravascular involvement, which occurred in women aged 73 and 48 years. Grossly and microscopically, both neoplasms had an extrauterine cotyledonoid part and intrauterine dissecting fascicles of disorganized, swirled neoplastic smooth muscle with hydropic degeneration and foci of an intravascular growth (the latter was identified histologically). To our knowledge, the intravascular component of such a neoplasm is a very rare feature that has previously been described only in three cases in the literature.