Uterine tumor resembling ovarian sex-cord tumor (UTROSCT) with sarcomatous features without recurrence after extended radical surgery

Abstract

Rationale: The malignant potential and the appropriate treatment of uterine tumor resembling ovarian sex-cord tumor (UTROSCT) is controversial. Although these tumors generally have benign outcomes, several reports have described recurrences, metastases, and deaths associated with this disease. Patient concerns: A 57-year-old Japanese woman (gravida 2, para 2) was referred to our hospital for the evaluation and treatment of uterine fibroids. Magnetic resonance imaging revealed a right ovarian mass and multiple fibroids in the uterine myometrium. Diagnoses: The patient was diagnosed with UTROSCT with sarcomatous features. Interventions: She initially underwent a total abdominal hysterectomy and bilateral salpingo-oophorectomy, followed by second-stage surgery comprising pelvic and para-aortic lymphadenectomy and subtotal omentectomy. Outcomes: No postoperative recurrence was observed in the patient in 36 months. Lessons: In this case, extended radical surgery prevented the development of recurrent disease in a patient with UTROSCT with sarcomatous features. These clinicopathological findings suggest that UTROSCT is associated with several risk factors, including older age, presence of necrosis, lymphovascular invasion, significant nuclear atypia, and significant mitotic activity. This lesion type should be considered malignant and treated with curative intent.

Uterine tumor resembling ovarian sex-cord tumor: case report and review of the literature

“…Jeong et al [ 16 ] reported a 32-year-old patient with UTROSCT and intramuscular infiltration who conceived after hysteroscopic resection of the tumor. Sato et al [ 4 ] reported a 57-year-old patient with UTROSCT and sarcoma features who initially underwent transabdominal total hysterectomy with bilateral adnexectomy, and then pelvic and abdominal paraaortic lymphadenectomy and subtotal omentectomy, based on postoperative pathology. The patient did not show recurrence within 36 months after surgery.…”

“…Type II UTROSCT is solely composed of sex-cord components and is usually benign or shows low-grade malignancy with occasional local recurrence or distant metastasis [ 2 ]. However, recent studies have reported postoperative recurrence and lymphatic metastasis in some cases of UTROSCT [ 3 , 4 ]. Some authorities proposed that UTROSCT should be categorized as a tumor with undetermined malignant potential.…”

Recurrent Uterine Tumor Resembling Ovarian Sex Cord Tumor: A Case report and Clinicopathological and Immunohistochemical Analysis

“…But the report emphasized the patients for conservative management should have no risk factors for recurrence and recommend careful follow-up. Miho Sato et al [33] reviewed the cases of UTROSCT with malignant behavior and concluded that a radical surgery including bilateral salpingo-oophorectomy, lymphadenectomy, and omentectomy might lead to a lower recurrence rate than a simple hysterectomy alone. Considering the adverse outcome of this case, we wonder whether giving a second-stage surgery or a radical surgery at that time might change the patient's prognosis.…”

Uterine Tumors Resembling Ovarian Sex Cord Tumors (UTROSCTs): A Scoping Review of 511 Cases, Including 2 New Cases

“…Elevated CA-125 (up to 2210 U/L in [100]) levels have been occasionally reported [60,88,100,135], linking with extrauterine tumor spread [60,100,110] or accompanying conditions like adenomyosis [135]. Notably, CA-125, HE4, and CEA, which were normal in some reports [66,77,111], are not typical markers for sex cord tumors. Interestingly, serum Inhibin levels, a marker for sex cord tumors, have not been reported, reflecting the oversight of sex cord differentiation during perioperative consideration.…”