Cotyledonoid Dissecting Leiomyoma of the Uterus with Intravascular Luminal Growth: A Case Study

Abstract

Here, we report the case of a 43-year-old female who was diagnosed with a cotyledonoid dissecting leiomyoma (CDL) of the uterus. CDL is a recently described and extremely rare variant of a benign leiomyoma that can grossly masquerade as a malignancy. The 13-cm sized tumor was located primarily on the extrauterine surface as an intrauterine continuity, which showed dark red, congested, bulbous protuberances. It was multinodular appearance, encasing the bilateral adnexae and the left iliac vein. Microscopically, the nodules were separated by extensive hydropic degeneration. The nodules were composed of cigar-shaped spindle cells with no mitosis, cellular pleomorphism or coagulation necrosis. They also showed an intravascular luminal growth pattern. CDL with intravascular growth was diagnosed after excluding intravascular leiomyomatosis, disseminated peritoneal leiomyomatosis, and benign metastasizing leiomyoma. The present case is the second reported case of CDL in Korea. Recognition of this rare and bizarre, malignancy-mimicking leiomyoma is crucial to prevent inappropriate treatment.

Cotyledonoid dissecting leiomyoma of the uterus: A report of four cases and a review of the literature

“…In the literature, to preserve fertility, the recommended treatment is resection of intrauterine tumors by myomectomy and extrauterine tumors by excision (1,5,12,13). In women of postmenopausal stage, total abdominal hysterectomy and bilateral salpingo-oophorectomy are recommended with removal of parametrically extended tumors (6,(14)(15)(16). No standard treatment for CDL has been identified; however, the prognosis of CDL is favorable.…”

“…At present, ~43 cases of CDL have been reported in the English literature (3). An accurate diagnosis of CDL is challenging prior to surgery, as the distinctive grapelike (4) gross appearance of an exophytic mass resembles placental tissue and is almost always misdiagnosed clinically as an ovarian tumor or uterine sarcoma (5)(6)(7)(8). The majority of patients with CDL are diagnosed during an exploratory laparotomy (1,(9)(10)(11), and a frozen section is recommended to be used for the diagnosis of CDL (7,9).…”

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.…”

“…An intravascular growth pattern has been reported in only one study (in 21.4% of cases) of CDL. [4] Intravascular leiomyomatosis has a benign outcome, although it can extend into the extra-uterine pelvic veins, inferior vena cava and even up to the right side of the heart. [4] The presence of intravascular growth could be misdiagnosed as a malignancy, such as leiomyosarcoma or endometrioid stromal sarcoma.…”

“…[4] Intravascular leiomyomatosis has a benign outcome, although it can extend into the extra-uterine pelvic veins, inferior vena cava and even up to the right side of the heart. [4] The presence of intravascular growth could be misdiagnosed as a malignancy, such as leiomyosarcoma or endometrioid stromal sarcoma. Unlike leiomyosarcomas, the smooth muscle cells in our case showed no cytological atypia, tumour cell necrosis nor increased mitotic activity.…”