Long‑term treatment of residual or recurrent low‑grade endometrial stromal sarcoma with aromatase inhibitors: A report of two cases and a review of the literature

Ryu,Hyewon; Choi,Yoon‑Seok; Song,Ik‑Chan; Yun,Hwan‑Jung; Jo,Deog‑Yeon; Kim,Samyong; Lee,Hyo  Jin

doi:10.3892/ol.2015.3674

Long‑term treatment of residual or recurrent low‑grade endometrial stromal sarcoma with aromatase inhibitors: A report of two cases and a review of the literature

Authors:
- Hyewon Ryu
- Yoon‑Seok Choi
- Ik‑Chan Song
- Hwan‑Jung Yun
- Deog‑Yeon Jo
- Samyong Kim
- Hyo Jin Lee
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Affiliations: Division of Hematology‑Oncology, Department of Internal Medicine, Chungnam National University Hospital, Daejeon 301‑721, Republic of Korea
Published online on: September 3, 2015 https://doi.org/10.3892/ol.2015.3674
Pages: 3310-3314

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Abstract

Endometrial stromal sarcoma (ESS) occurs rarely and accounts for only 0.2% of all uterine malignancies. ESS usually expresses estrogen and progesterone receptors, and is regarded as hormone‑sensitive. Due to the rarity of these tumors, there are only few case series on the use of aromatase inhibitors in the treatment of low‑grade ESS. The present study reports the cases of two patients with residual or recurrent low‑grade ESS who experienced long‑term disease‑free survival following treatment with letrozole. The study also reviews the literature with regard to the data on aromatase inhibitors used in patients with low‑grade ESS. In total, 30 patients with recurrent or residual low‑grade ESS who were treated with aromatase inhibitors were identified, including the present cases. Among the 30 patients, the overall response rate of advanced low‑grade ESS to aromatase inhibitors was 77.4% (complete response, 25.8%; partial response, 51.6%) and the disease control rate was 90.3%. The response rate of first‑line treatment was similar to that of second‑line therapy or higher (84.6 vs. 72.2%; P=0.453). Duration of aromatase inhibitor treatment ranged from 1.5 to 168 months (median, 26.5 months). The aromatase inhibitors showed minimal adverse effects. In conclusion, aromatase inhibitors, particularly third‑generation drugs, are a well‑tolerated class of medications that are effective in the treatment of advanced low‑grade ESS, with a favorable toxicity profile.

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1	Olah KS, Gee H, Blunt S, Dunn JA, Kelly K and Chan KK: Retrospective analysis of 318 cases of uterine sarcoma. Eur J Cancer. 27:1095–1099. 1991. View Article : Google Scholar : PubMed/NCBI
2	Harlow BL, Weiss NS and Lofton S: The epidemiology of sarcomas of the uterus. J Natl Cancer Inst. 76:399–402. 1986.PubMed/NCBI
3	Evans HL: Endometrial stromal sarcoma and poorly differentiated endometrial sarcoma. Cancer. 50:2170–2182. 1982. View Article : Google Scholar : PubMed/NCBI
4	Amant F, Vergote I and Moerman P: The classification of a uterine sarcoma as ‘high-grade endometrial stromal sarcoma’ should be abandoned. Gynecol Oncol. 95:412–413. (author reply 413). 2004.PubMed/NCBI
5	Reich O and Regauer S: Survey of adjuvant hormone therapy in patients after endometrial stromal sarcoma. Eur J Gynaecol Oncol. 27:150–152. 2006.PubMed/NCBI
6	Pink D, Lindner T, Mrozek A, Kretzschmar A, Thuss-Patience PC, Dörken B and Reichardt P: Harm or benefit of hormonal treatment in metastatic low-grade endometrial stromal sarcoma: Single center experience with 10 cases and review of the literature. Gynecol Oncol. 101:464–469. 2006. View Article : Google Scholar : PubMed/NCBI
7	Ashraf-Ganjoei T, Behtash N, Shariat M and Mosavi A: Low grade endometrial stromal sarcoma of uterine corpus, a clinico-pathological and survey study in 14 cases. World J Surg Oncol. 4:502006. View Article : Google Scholar : PubMed/NCBI
8	Amant F, Moerman P, Cadron I, Neven P, Berteloot P and Vergote I: The diagnostic problem of endometrial stromal sarcoma: Report on six cases. Gynecol Oncol. 90:37–43. 2003. View Article : Google Scholar : PubMed/NCBI
9	Reich O and Regauer S: Hormonal therapy of endometrial stromal sarcoma. Curr Opin Oncol. 19:347–352. 2007. View Article : Google Scholar : PubMed/NCBI
10	Therasse P, Arbuck SG, Eisenhauer EA, Wanders J, Kaplan RS, Rubinstein L, Verweij J, Van Glabbeke M, van Oosterom AT, Christian MC and Gwyther SG: New guidelines to evaluate the response to treatment in solid tumors. European Organization for Research and Treatment of Cancer, National Cancer Institute of the United States, National Cancer Institute of Canada. J Natl Cancer Inst. 92:205–216. 2000. View Article : Google Scholar : PubMed/NCBI
11	Maluf FC, Sabbatini P, Schwartz L, Xia J and Aghajanian C: Endometrial stromal sarcoma: Objective response to letrozole. Gynecol Oncol. 82:384–388. 2001. View Article : Google Scholar : PubMed/NCBI
12	Spano JP, Soria JC, Kambouchner M, Piperno-Neuman S, Morin F, Morere JF, Martin A and Breau JL: Long-term survival of patients given hormonal therapy for metastatic endometrial stromal sarcoma. Med Oncol. 20:87–93. 2003. View Article : Google Scholar : PubMed/NCBI
13	Leunen M, Breugelmans M, De Sutter P, Bourgain C and Amy JJ: Low-grade endometrial stromal sarcoma treated with the aromatase inhibitor letrozole. Gynecol Oncol. 95:769–771. 2004. View Article : Google Scholar : PubMed/NCBI
14	Leiser AL, Hamid AM and Blanchard R: Recurrence of prolactin-producing endometrial stromal sarcoma with sex-cord stromal component treated with progestin and aromatase inhibitor. Gynecol Oncol. 94:567–571. 2004. View Article : Google Scholar : PubMed/NCBI
15	Brechot JM, Kamboucher M, Brauner M, Destable M, Duperron F and Morère JF: Pulmonary metastases from endometrial stromal sarcoma may benefit from hormone therapy. Rev Mal Respir. 24:69–72. 2007.(In French). PubMed/NCBI
16	Landréat V, Paillocher N, Catala L, Foucher F, Descamps P and Levêque J: Low-grade endometrial stromal sarcoma of the uterus: Review of 10 cases. Anticancer Res. 28:2869–2874. 2008.PubMed/NCBI
17	Krauss K, Bachmann C, Hartmann JT, Siegmann K, Sotlar K, Wallwiener D and Huober J: Management of late recurrence of a low-grade endometrial stromal sarcoma (LGESS): Treatment with letrozole. Anticancer Res. 27:3477–3480. 2007.PubMed/NCBI
18	Alkasi O, Meinhold-Heerlein I, Zaki R, Fasching P, Maass N, Jonat W and Beckmann MW: Long-term disease-free survival after hormonal therapy of a patient with recurrent low grade endometrial stromal sarcoma: A case report. Arch Gynecol Obstet. 279:57–60. 2009. View Article : Google Scholar : PubMed/NCBI
19	Tzakas E, Liu S, Todd RW and Redman CW: Hormonal therapy with letrozole prior to surgical management of recurrent metastatic low-grade endometrial stromal sarcoma (LGESS). J Obstet Gynaecol. 29:778–779. 2009. View Article : Google Scholar : PubMed/NCBI
20	Dahhan T, Fons G, Buist MR, Ten Kate FJ and van der Velden J: The efficacy of hormonal treatment for residual or recurrent low-grade endometrial stromal sarcoma. A retrospective study. Eur J Obstet Gynecol Reprod Biol. 144:80–84. 2009. View Article : Google Scholar : PubMed/NCBI
21	Ioffe YJ, Li AJ, Walsh CS, Karlan BY, Leuchter R, Forscher C and Cass I: Hormone receptor expression in uterine sarcomas: Prognostic and therapeutic roles. Gynecol Oncol. 115:466–471. 2009. View Article : Google Scholar : PubMed/NCBI
22	Sylvestre VT and Dunton CJ: Treatment of recurrent endometrial stromal sarcoma with letrozole: A case report and literature review. Horm Cancer. 1:112–115. 2010. View Article : Google Scholar : PubMed/NCBI
23	Shoji K, Oda K, Nakagawa S, Kawana K, Yasugi T, Ikeda Y, Takazawa Y, Kozuma S and Taketani Y: Aromatase inhibitor anastrozole as a second-line hormonal treatment to a recurrent low-grade endometrial stromal sarcoma: A case report. Med Oncol. 28:771–774. 2011. View Article : Google Scholar : PubMed/NCBI
24	Altman AD, Nelson GS, Chu P, Nation J and Ghatage P: Uterine sarcoma and aromatase inhibitors: Tom baker cancer centre experience and review of the literature. Int J Gynecol Cancer. 22:1006–1012. 2012. View Article : Google Scholar : PubMed/NCBI
25	Balleine RL, Earls PJ, Webster LR, Mote PA, de Fazio A, Harnett PR and Clarke CL: Expression of progesterone receptor A and B isoforms in low-grade endometrial stromal sarcoma. Int J Gynecol Pathol. 23:138–144. 2004. View Article : Google Scholar : PubMed/NCBI
26	Chu MC, Mor G, Lim C, Zheng W, Parkash V and Schwartz PE: Low-grade endometrial stromal sarcoma: Hormonal aspects. Gynecol Oncol. 90:170–176. 2003. View Article : Google Scholar : PubMed/NCBI
27	Reich O, Regauer S, Urdl W, Lahousen M and Winter R: Expression of oestrogen and progesterone receptors in low-grade endometrial stromal sarcomas. Br J Cancer. 82:1030–1034. 2000.PubMed/NCBI