J Gynecol Oncol.  2015 Jul;26(3):214-221. 10.3802/jgo.2015.26.3.214.

Long-term survival of patients with recurrent endometrial stromal sarcoma: a multicenter, observational study

• Yamazaki H
• Todo Y
• Mitsube K
• Hareyama H
• Shimada C
• Kato H
• Yamashiro

• KMID: 2160808
• DOI: http://doi.org/10.3802/jgo.2015.26.3.214

Abstract

OBJECTIVE
The aim of this study was to evaluate the clinical behavior and management outcome of recurrent endometrial stromal sarcoma (ESS).
METHODS
A retrospective review of charts of 10 patients with recurrent ESS was performed and relapse-free interval, relapse site, treatment, response to treatment, duration of follow-up and clinical outcome extracted. Survival outcome measures used were post-relapse survival which was defined as the time from first evidence of relapse to death from any cause. Living patients were censored at the date of last follow-up.
RESULTS
The median age and median relapse-free interval at the time of initial relapse were 51.5 years and 66.5 months, respectively. The number of relapses ranged from one to five. Sixteen surgical procedures for recurrent disease included nine (56.0%) complete resections. There was no statistically significant difference between initial recurrent tumors and second/subsequent recurrent tumors in the rate of complete surgery (44.4% vs. 71.4%, respectively, p=0.36). Of the eleven evaluable occasions when hormonal therapy was used for recurrent disease, disease control was achieved in eight (72.7%). There was no difference between initial recurrent tumors and second/subsequent recurrent tumors in disease control rate by hormonal therapy (85.7% vs. 50.0%, respectively, p=0.49). The 10-year post-relapse survival rate was 90.0% and the overall median post-relapse survival 119 months (range, 7 to 216 months).
CONCLUSION
Post-relapse survival of patients with ESS can be expected to be >10 years when treated by repeated surgical resection and hormonal therapy or both.

Keyword

Aromatase Inhibitors; Gonadotropin-Releasing Hormone; Retrospective Studies; Sarcoma, Endometrial Stromal; Survival Rate

MeSH Terms

Adult
Aged
Antineoplastic Agents, Hormonal/therapeutic use
Chemotherapy, Adjuvant/mortality
Disease-Free Survival
Endometrial Neoplasms/drug therapy/*mortality/surgery
Female
Humans
Middle Aged
Neoplasm Recurrence, Local/*mortality
Retrospective Studies
Sarcoma, Endometrial Stromal/drug therapy/*mortality/surgery
Treatment Outcome
Antineoplastic Agents, Hormonal

Figure

• 

  Fig. 1 (A) Relapse-free interval prior to initial relapse and (B) post-relapse survival curves for patients with recurrent endometrial stromal sarcoma.

Reference

1. Hendrickson MR, Tavassoli FA, Kempson RL, McCluggage WG, Haller U, Kubik-Huch RA. Mesenchymal tumours and related lesions. In : Tavassoli FA, Devilee P, editors. World Health Organization classification of tumours. Pathology and genetics of tumours of the breast and female genital organs. Lyon: IARC Press;2003. p. 233–244.

2. Boll D, Verhoeven RH, van der Aa MA, Pauwels P, Karim-Kos HE, Coebergh JW, et al. Incidence and survival trends of uncommon corpus uteri malignancies in the Netherlands, 1989-2008. Int J Gynecol Cancer. 2012; 22:599–606.

3. Norris HJ, Taylor HB. Mesenchymal tumors of the uterus. I. A clinical and pathological study of 53 endometrial stromal tumors. Cancer. 1966; 19:755–766.

4. Krieger PD, Gusberg SB. Endolymphatic stromal myosis: a grade I endometrial sarcoma. Gynecol Oncol. 1973; 1:299–313.

5. Piver MS, Rutledge FN, Copeland L, Webster K, Blumenson L, Suh O. Uterine endolymphatic stromal myosis: a collaborative study. Obstet Gynecol. 1984; 64:173–178.

6. Mansi JL, Ramachandra S, Wiltshaw E, Fisher C. Endometrial stromal sarcomas. Gynecol Oncol. 1990; 36:113–118.

7. Berchuck A, Rubin SC, Hoskins WJ, Saigo PE, Pierce VK, Lewis JL Jr. Treatment of endometrial stromal tumors. Gynecol Oncol. 1990; 36:60–65.

8. Larson B, Silfversward C, Nilsson B, Pettersson F. Endometrial stromal sarcoma of the uterus. A clinical and histopathological study. The Radiumhemmet series 1936-1981. Eur J Obstet Gynecol Reprod Biol. 1990; 35:239–249.

9. Goff BA, Rice LW, Fleischhacker D, Muntz HG, Falkenberry SS, Nikrui N, et al. Uterine leiomyosarcoma and endometrial stromal sarcoma: lymph node metastases and sites of recurrence. Gynecol Oncol. 1993; 50:105–109.

10. Gadducci A, Sartori E, Landoni F, Zola P, Maggino T, Urgesi A, et al. Endometrial stromal sarcoma: analysis of treatment failures and survival. Gynecol Oncol. 1996; 63:247–253.

11. Chu MC, Mor G, Lim C, Zheng W, Parkash V, Schwartz PE. Low-grade endometrial stromal sarcoma: hormonal aspects. Gynecol Oncol. 2003; 90:170–176.

12. Li AJ, Giuntoli RL 2nd, Drake R, Byun SY, Rojas F, Barbuto D, et al. Ovarian preservation in stage I low-grade endometrial stromal sarcomas. Obstet Gynecol. 2005; 106:1304–1308.

13. Leath CA 3rd, Huh WK, Hyde J Jr, Cohn DE, Resnick KE, Taylor NP, et al. A multi-institutional review of outcomes of endometrial stromal sarcoma. Gynecol Oncol. 2007; 105:630–634.

14. Amant F, De Knijf A, Van Calster B, Leunen K, Neven P, Berteloot P, et al. Clinical study investigating the role of lymphadenectomy, surgical castration and adjuvant hormonal treatment in endometrial stromal sarcoma. Br J Cancer. 2007; 97:1194–1199.

15. Nam EJ, Kim JW, Lee DW, Jang SY, Hong JW, Kim YT, et al. Endometrial stromal sarcomas: a retrospective analysis of 28 patients, single center experience for 20 years. Cancer Res Treat. 2008; 40:6–10.

16. Kim WY, Lee JW, Choi CH, Kang H, Kim TJ, Kim BG, et al. Low-grade endometrial stromal sarcoma: a single center's experience with 22 cases. Int J Gynecol Cancer. 2008; 18:1084–1089.

17. Beck TL, Singhal PK, Ehrenberg HM, Rose PG, Lele SB, Krivak TC, et al. Endometrial stromal sarcoma: analysis of recurrence following adjuvant treatment. Gynecol Oncol. 2012; 125:141–144.

18. Mizuno M, Yatabe Y, Nawa A, Nakanishi T. Long-term medroxyprogesterone acetate therapy for low-grade endometrial stromal sarcoma. Int J Clin Oncol. 2012; 17:348–354.

19. Yoon A, Park JY, Park JY, Lee YY, Kim TJ, Choi CH, et al. Prognostic factors and outcomes in endometrial stromal sarcoma with the 2009 FIGO staging system: a multicenter review of 114 cases. Gynecol Oncol. 2014; 132:70–75.

20. Rauh-Hain JA, del Carmen MG. Endometrial stromal sarcoma: a systematic review. Obstet Gynecol. 2013; 122:676–683.

21. Gadducci A, Cosio S, Romanini A, Genazzani AR. The management of patients with uterine sarcoma: a debated clinical challenge. Crit Rev Oncol Hematol. 2008; 65:129–142.

22. Reich O, Regauer S. Hormonal therapy of endometrial stromal sarcoma. Curr Opin Oncol. 2007; 19:347–352.

23. Pink D, Lindner T, Mrozek A, Kretzschmar A, Thuss-Patience PC, Dorken B, et al. 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. 2006; 101:464–469.

24. Maluf FC, Sabbatini P, Schwartz L, Xia J, Aghajanian C. Endometrial stromal sarcoma: objective response to letrozole. Gynecol Oncol. 2001; 82:384–388.

25. Krauss K, Bachmann C, Hartmann JT, Siegmann K, Sotlar K, Wallwiener D, et al. Management of late recurrence of a low-grade endometrial stromal sarcoma (LGESS): treatment with letrozole. Anticancer Res. 2007; 27(5B):3477–3480.

26. Mesia AF, Demopoulos RI. Effects of leuprolide acetate on low-grade endometrial stromal sarcoma. Am J Obstet Gynecol. 2000; 182:1140–1141.

27. Burke C, Hickey K. Treatment of endometrial stromal sarcoma with a gonadotropin-releasing hormone analogue. Obstet Gynecol. 2004; 104(5 Pt 2):1182–1184.

28. Weitmann HD, Knocke TH, Kucera H, Potter R. Radiation therapy in the treatment of endometrial stromal sarcoma. Int J Radiat Oncol Biol Phys. 2001; 49:739–748.

29. Sutton G, Blessing JA, Park R, DiSaia PJ, Rosenshein N. Ifosfamide treatment of recurrent or metastatic endometrial stromal sarcomas previously unexposed to chemotherapy: a study of the Gynecologic Oncology Group. Obstet Gynecol. 1996; 87(5 Pt 1):747–750.

30. Jordan VC. Medroxyprogesterone acetate and metastases: of mice and (wo)men. J Natl Cancer Inst. 2005; 97:619–621.

31. Thigpen JT, Brady MF, Alvarez RD, Adelson MD, Homesley HD, Manetta A, et al. Oral medroxyprogesterone acetate in the treatment of advanced or recurrent endometrial carcinoma: a dose-response study by the Gynecologic Oncology Group. J Clin Oncol. 1999; 17:1736–1744.

32. Sugimoto AK, Hodsman AB, Nisker JA. Long-term gonadotropin-releasing hormone agonist with standard postmenopausal estrogen replacement failed to prevent vertebral bone loss in premenopausal women. Fertil Steril. 1993; 60:672–674.

33. Paoletti AM, Serra GG, Cagnacci A, Vacca AM, Guerriero S, Solla E, et al. Spontaneous reversibility of bone loss induced by gonadotropin-releasing hormone analog treatment. Fertil Steril. 1996; 65:707–710.

34. Fontein DB, Nortier JW, Liefers GJ, Putter H, Meershoek-Klein Kranenbarg E, van den Bosch J, et al. High non-compliance in the use of letrozole after 2.5 years of extended adjuvant endocrine therapy. Results from the IDEAL randomized trial. Eur J Surg Oncol. 2012; 38:110–117.

35. Reerink EH, Scholer HF, Westerhof P, Querido A, Kassenaar AA, Diczfalusy E, et al. A new class of hormonally active steroids. Nature. 1960; 186:168–169.

36. Schindler AE. Progestational effects of dydrogesterone in vitro, in vivo and on the human endometrium. Maturitas. 2009; 65:Suppl 1. S3–S11.

37. Akashi D, Todo Y, Shimada C, Okamoto K, Minobe S, Kato H. Successful use of dydrogesterone as maintenance therapy in recurrent endometrial stromal sarcoma: a case report. Jpn J Clin Oncol. 2013; 43:1145–1149.

38. Chan JK, Kawar NM, Shin JY, Osann K, Chen LM, Powell CB, et al. Endometrial stromal sarcoma: a population-based analysis. Br J Cancer. 2008; 99:1210–1215.

39. Shah JP, Bryant CS, Kumar S, Ali-Fehmi R, Malone JM Jr, Morris RT. Lymphadenectomy and ovarian preservation in low-grade endometrial stromal sarcoma. Obstet Gynecol. 2008; 112:1102–1108.