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Background: The role of lymphadenectomy in low-grade endometrial stromal sarcoma (ESS) is controversial. The risk of nodal metastases ranges from 0% to 44%, but data are inconclusive. The objective of this study and of the literature review was to investigate the incidence of nodal involvement in macroscopically early-stage tumors. Methods: All consecutive patients with histologically proven uterine low-grade ESS who underwent surgery in our institution were considered eligible for the analysis. Until July 2006, pelvic systematic lymphadenectomy was performed based on the physician's choice, whereas after July 2006, all women with apparent early-stage tumor underwent systematic pelvic nodes dissection. Results: From January 1985 to March 2009, 64 women were surgically treated and 19 underwent bilateral systematic pelvic lymphadenectomy. The median count of nodes removed was 20 (range, 11-30). Nodal involvement was depicted in 3 of 19 women but only in 1 in 16 (5%) women with apparent early-stage disease. Specifically, we did not find nodal metastases in 9 women with macroscopically early-stage tumors treated after July 2006. According to the literature review, the risk of nodal metastases is 5% (range, 0%-16%) in early-stage disease. Conclusions: There is no indication to offer a systematic lymphadenectomy in apparent early-stage low-grade ESS according to our analysis and to the literature review. Retroperitoneal surgery should be limited in case of bulky nodal disease or in case of nodal recurrences