Lipoleiomyoma is a rare, benign leiomyoma variant. It is relatively common in the uterine area of the female reproductive system but rare in other areas and has not been reported in the fallopian tubes. In this paper, we report a perimenopausal woman with a lipoleiomyoma arising from the ampulla of the fallopian tube with hydropic degeneration. What makes this case even rarer is the combination of hydrops of the pelvis and the abdominal cavity. Microscopically, the tumor was composed of smooth muscle tissue mixed with varying amounts of mature adipose tissue. The immunohistochemical markers were Des(+), SMA(+), H-caldesmon(+), ER(+), CD34(-), HMB45(-), Melan-A(-), CD10(-), S-100 focal adipocyte (+), and the positive rate of Ki67 was about 1%. Through this case report and review of similar literature, we hope to improve the understanding of the diagnosis and treatment of fallopian tube smooth muscle-derived tumors.