An unusual case of uterine cotyledonoid dissecting leiomyoma with adenomyosis

Abstract

BackgroundCotyledonoid dissecting leiomyoma is a rare variant of uterine smooth muscle tumor with an unusual growth pattern that shows intramural dissection within uterine myometrium and often a placenta-like appearance in its extrauterine components.Case presentationWe present a unique case of cotyledonoid dissecting leiomyoma with adenomyosis. A 40-year-old Japanese female presented with prolonged menorrhagia and severe anemia. She had a pelvic mass followed-up for 6 years with a diagnosis of leiomyoma. However, increase in tumor size and cystic changes with hemorrhage were found by magnetic resonance imaging, and total abdominal hysterectomy with bilateral salpingectomy was performed. Macroscopically, the placenta-like exophytic mass protruding from the posterior uterine wall was composed of multiple nodules containing numerous hemorrhagic cysts. The mass showed continuity as a white multinodular dissecting mass infiltrating the posterolateral myometrium. Microscopically, both extra–and intrauterine portions of the mass were composed of nodules that contained swirled neoplastic smooth muscle cells with marked hyalinized degeneration, as observed in cotyledonoid dissecting leiomyomas of conventional type. In addition, numerous non–neoplastic glands of endometrial type surrounded by abundant endometrium–like stromal cells and non–neoplastic smooth muscle cells were found in the tumor, suggesting that it involved a part of concomitant adenomyosis originating from the nontumoral myometrium.ConclusionsThus far, over 30 cases of cotyledonoid dissecting leiomyoma have been reported, none of which have described the presence of adenomyosis within the tumor. The present case suggested that cotyledonoid dissecting leiomyoma might have a unique clinical presentation involving concomitant uterine adenomyosis. It is critical for pathologists, gynecologists, and radiologists to be cognizant of cotyledonoid dissecting leiomyoma variants for timely and appropriate diagnosis and treatment.

Cotyledonoid Leiomyoma Clinical Characteristics, Imaging Features, and Review of the Literature

“…Some special CL cases were also described in the literature. Microscopically, it may even arise from a part of an adenomyotic myometrium 3,28 or may englobe some endometrial tissue organized in hemorrhagic cysts 28 . Endosalpingiosis has also been associated with uterine CDL as an incidental finding on macroscopic and microscopic examinations (unique case from Driss et al 29 ).…”

Cotyledonoid Dissecting Leiomyoma of the Uterus: An Unexpected Diagnosis After Delivery

“…Uterine smooth muscle tumors are known to exhibit a wide variety of growth patterns, with the unusual variants accounting for approximately 10%. 3 One rare variant is the cotyledonoid dissecting leiomyoma (CDL), initially recognized in 1975 and described by Roth et al in 1996 as a tumor that presents clinically and radiologically as an apparent malignant growth. 4 The age distribution ranges from 23 to 73 years.…”

“…Other interesting fact is that several reports alluded to CDL with non-neoplastic cyst lesions, such as adenomyosis, 3 adenoleiomyomatous component, 3 limited foci of endometriosis 8 and endosalpingiosis. 9 These cases suggest that CDL might have a unique clinical presentation involving parts of benign non-neoplastic cystic lesions.…”

“…The most common treatment for this type of tumor to date has been hysterectomy, influenced by its macroscopic appearance. Roth et al, 5 Saeki et al 3 and Kim et al 10 reported cases where myomectomy with resection of the extrauterine tumor was performed. One of those women, 2 a year after the surgery, became pregnant naturally.…”

Cotyledonoid dissecting leiomyoma with intravascular growth pattern and intra-tumoural endometrial glands and stroma: A case report

“…Over 30 cases of this tumour have been reported up to this date in the published literature, one of which had described the presence of adenomyosis within the tumour. [3] Uterine cotyledonoid dissecting leiomyomas with intravascular luminal growth, [4] epithelioid variant, [5] hydropic variant [6] and CDL with adipocytic differentiation [7] have also been described. The present case is the second described case in the published literature of a CDL in which non-neoplastic endometrial glands with endometrial stromal cells are present together with an intravascular luminal growth component.…”