Hydatidiform mole is a pregnancy disorder, of a benign nature. We present a case of molar tissue within a uterine myoma, the first such entity reported in the literature. In May 2006, a thirty-eight year old infertility patient was admitted for myomectomy. She had anamnesis for chronic pelvic inflammatory disease and surgeries performed for tubal pregnancies. After the sixth intracytoplasmal spermatozoa injection procedure performed in January 2006, she conceived, but curettage was performed in March 2006 for a missed abortion. Following the routine preoperative evaluation in May 2006, four months after the last artificial reproductive technology procedure, myomectomy was performed as uneventful operation, but the histological report appeared unusual, showing degenerated chorionic villi within the uterine myoma. Molar tissue within uterine myoma might evolve even after artificial reproductive procedures. Furthermore, this finding might be misinterpreted as a fibromyoma degeneration. This is the first, and a unique case, of molar tissue within uterine myoma reported in the literature.