Fertility-sparing management of low-grade endometrial stromal sarcoma: analysis of an institutional series, a population-based analysis and review of the literature

Abstract

Background: Low-grade endometrial stromal sarcoma (LGESS) is the second most common malignant mesenchymal tumor of the uterus which usually affects young women. However, the researches on the safety and feasibility of the fertility-sparing management of it are limited.Methods: A retrospective analysis was performed including 5 women diagnosed with LGESS treated with fertility-sparing management at Qilu Hospital of Shandong University from 2010 to 2019. Besides that, 1,070 patients diagnosed with LGESS in SEER database from 1973 to 2016 were examined. By using the Kaplan-Meier method, survival curves were estimated, and comparisons of statistical significance were performed with the stratified log-rank test within each group.Results: Five patients with LGESS were enrolled in this study. All patients were submitted to fertilitysparing surgeries, after surgery, they all continued hormonal therapy for one year. Four out of the 5 patients recurred, to be more exact, 3 of them recurred in uterus and the other one in the uterus and iliac vascular region. They all suffered further surgery and all 5 patients were alive at the time of last contact. Besides, among these patients, two conceived naturally and delivered a healthy baby by cesarean section. Among 1,070 patients in SEER database, only 28 (2.6%) patients underwent local tumor excision, including excisional biopsy (39%), myomectomy (25%), laser ablation or excision (4%) and polypectomy (4%). There was no statistical significance was observed among TH±BSO, radical hysterectomy, subtotal hysterectomy and local tumor excision (P=0.29).Conclusions: Our analysis indicated that for those young LGESS patients who wish to preserve their fertility, the feasibility and safety of fertility-sparing management should be considered after gynecological oncologist and gynecological pathologist making professional decisions.

Hormone Therapy Reduces Recurrence in Stage II-IV Uterine Low-Grade Endometrial Stromal Sarcomas: A Retrospective Cohort Study

“…The proportion of patients receiving postoperative hormone therapy was 69.5% in our study, whereas it was as high as 83.3%-100% in other studies ( 29 – 31 ). However, our cohort included a larger number of patients compared to these studies.…”

“…Most patients underwent lesion resection by laparoscopy or laparotomy, possibly because myoma was the most common clinical presentation. The proportion of patients receiving postoperative hormone therapy was 69.5% in our study, whereas it was as high as 83.3%-100% in other studies (29)(30)(31). However, our cohort included a larger number of patients compared to these studies.…”

“…Except for high-dose progestins, GnRH-a was a relatively common choice of hormone therapy in the fertility-sparing cohort; this was quite different from the cohort without fertility-sparing. The most common duration of hormone therapy was 6 months or less, which was shorter than that described in other studies (30)(31)(32). Hormone therapy seemed to prolong DFS in these patients, although this effect was not significant.…”

“…Among these patients, five received hormone therapy, including high-dose progestins and/or GnRH-a. In the few reports so far, patients with successful delivery used only progestins for hormone therapy (29)(30)(31)(32). Thus, our findings suggest that GnRH-a is also a good option for fertility-sparing hormone therapy.…”

A difficult to diagnose case of low‑grade endometrial stromal sarcoma with smooth muscle differentiation treated with laparoscopic surgery: A case report

“…It is difficult to distinguish between LGESS and other types of tumors, such as leiomyoma with degeneration or cellular leiomyoma by MRI examination before surgery. Therefore, it is not uncommon for LGESS to be identified by postoperative pathological diagnosis after hysterectomy or tumorectomy for preoperative diagnosis of uterine leiomyoma, and there are reports of LGESS being diagnosed during/after laparoscopic surgery (6)(7)(8). When laparoscopic surgery is performed on LGESS, the risk of intraperitoneal dissemination should be taken into consideration.…”