Medical Oncology

, 20:87 | Cite as

Long-term survival of patients given hormonal therapy for metastatic endometrial stromal sarcoma

  • J. -P. Spano
  • J. C. Soria
  • M. Kambouchner
  • S. Piperno-Neuman
  • F. Morin
  • J. -F. Morere
  • A. Martin
  • J. -L Breau
Case Report

Abstract

Endometrial stromal sarcoma (ESS) is a rare neoplasm, mainly observed in premenopausal women. We describe two women 44 and 34 years old at the time ESS diagnosis, who developed lung metastases 3 and 6 years, respectively, after initial treatment: hysterectomy without (case 1) or with oophorectomy (case 2), followed by hormone replacement therapy (HRT) for the latter. Their estrogen (ER) and progesterone receptors (PR) were analyzed biochemically in metastatic lung tissue, yielding respective concentrations of ER 242 and 184, and PR 910 and 100 fmol/mg of cytosol protein. Both patients started treatment with the aromatase inhibitor aminoglutethimide (500 mg qid) after surgery for the first patient and after stopping HRT for the second. Under aromatase-inhibitor therapy, both patients achieved a complete response, patient 1 remains disease-free with 14+ years of follow-up, and patient 2 with 7+ years. Our data suggest that an aromatase inhibitor may be an effective treatment for ESS. Furthermore, routine ER and PR analyses could be useful to predict the response to hormonal therapy in ESS.

Key Words

Aromatase inhibitor estrogen receptor progesterone receptor uterine stromal sarcoma 

References

  1. 1.
    Boutselis, J.G. and Ullery, J.C. (1962). Sarcomas of the uterus. Obstet Gynecol 20:23–35.PubMedCrossRefGoogle Scholar
  2. 2.
    Yoonessi, M. and Hart, W.R. (1977) Endometrial stromal sarcomas. Cancer 40:898–906.PubMedCrossRefGoogle Scholar
  3. 3.
    Koss, L.G., Spiro, R.H. and Brunschwig, A. (1965) Endometrial stromal sarcoma. Surg Gynecol Obstet 121:531–537.PubMedGoogle Scholar
  4. 4.
    Katz, L., Merino, M.J., Sakamoto, H. and Schwartz, P. (1987) Endometrial stromal sarcoma: a clinicopathologic study of 11 cases with determination of estrogen and progestin receptor levels in three tumors. Gynecol Oncol 26:87–97.PubMedCrossRefGoogle Scholar
  5. 5.
    Tsukamoto, N., Kamura, T. and Mutsukuma, K. (1985) Endolymphatic stromal myosis: a case with positive estrogen and progesterone receptors and good response to progestins. Gynecol Oncol 20:120–128.PubMedCrossRefGoogle Scholar
  6. 6.
    Scribner, D.R. and Walker, J.L. (1998) Low grade endometrial stromal sarcoma preoperative treatment with Depo-Lupron and megace. Gynecol Oncol 71(3):458–460.PubMedCrossRefGoogle Scholar
  7. 7.
    Maluf, F.C., Sabbatini, P., Schwartz, L., Xia, J. and Aghajanian, C. (2001) Endometrial stromal sarcoma: objective response to letrozole. Gynecol Oncol 82:384–388.PubMedCrossRefGoogle Scholar
  8. 8.
    Stuart-Harris, R.C. and Smith, I.E. (1984) Aminoglutethimide in the treatment of advanced breast cancer. Cancer Treat Rev 11:189–204.PubMedCrossRefGoogle Scholar
  9. 9.
    Buzdar, A. and Howell, A. (2001) Advances in aromatase inhibition: clinical efficacy and tolerability in the treatment of breast cancer. Clin Cancer Research 7:2620–2635.Google Scholar
  10. 10.
    Doran, A.H.G. and Lockyer, C. (1908) Two cases of uterine fibroids sharing peritheliomatous changes: long immunity from recurrence after operation. Proc R Soc Med 25:2.Google Scholar
  11. 11.
    Hart, W.R. and Yoonessi, M. (1977) Endometrial stromatosis of the uterus, Obstet Gynecol 49:393–403.PubMedGoogle Scholar
  12. 12.
    Kerby, I.J. (1975) Stromal endometriosis. Clin Radiol 26:99–101.PubMedCrossRefGoogle Scholar
  13. 13.
    Barlesi, F., et al. (2001) Multiple pulmonary leiomyomatous nodules: benign metastasizing leiomyoma? Rev Mal Respir 18:440–442.PubMedGoogle Scholar
  14. 14.
    Jautzke, G., Muller-Ruchholtz, E. and Thalmann, U. (1996) Immunohistological detection of estrogen and progesterone receptors in multiple and well-differentiated leiomyomatous lung tumors in women with uterine leiomyomas (so-called benign metastasizing leiomyomas). A report on 5 cases. Pathol Res Pract 192:215–223.PubMedGoogle Scholar
  15. 15.
    Canzonieri, V., et al. (1994) Leiomyomatosis with vascular invasion. A unified pathogenesis regarding leiomyoma with vascular microinvasion, benign metastasizing leiomyoma and intravenous leiomyomatosis. Virschows Arch 425:541–545.Google Scholar
  16. 16.
    McCluggage, W.G., Bailie, C., Weir, P. and Bharucha, H. (1996) Endometrial stromal sarcoma arising in pelvic endometriosis in a patient receiving unopposed oestrogen therapy. Br J Obstet Gynaecol 103:1252–1254.PubMedGoogle Scholar
  17. 17.
    Ursic-Vrscaj, M. (1999) Hormone replacement therapy after uterine leiomyosarcoma treatment. Case reports. Eur J Gynaecal Oncol 20:379–382.Google Scholar
  18. 18.
    Pellilo, D. (1968) Proliferative stromatosis of the uterus with pulmonary metastases. Remission following treatment with a long-acting synthetic progestin: a case report. Obstet Gynecol 49:393–403.Google Scholar
  19. 19.
    Tseng, L., et al. (1986) Endocrine aspects of human uterine sarcoma: a preliminary study. Am J Obstet Gynecol 155:95–101.PubMedGoogle Scholar
  20. 20.
    Schwartz, P.E. (1983) Steroid-receptor proteins in nonepithelial malignancies of the ovary. Gynecol Oncol 15:305–315.PubMedCrossRefGoogle Scholar
  21. 21.
    Dowbernowsky, P., Smith, I. and Falkson, G. (1998) Letrozole (Femara), a new oral aromatase inhibitor for advanced breast cancer: double-blind randomized trial showing a dose effect and improved efficacy and tolerability compared with megestrol acetate. J Clin Oncol 16:453–461.Google Scholar

Copyright information

© Humana Press Inc 2003

Authors and Affiliations

  • J. -P. Spano
    • 1
  • J. C. Soria
    • 2
  • M. Kambouchner
    • 3
  • S. Piperno-Neuman
    • 4
  • F. Morin
    • 1
  • J. -F. Morere
    • 1
  • A. Martin
    • 3
  • J. -L Breau
    • 1
  1. 1.Department of OncologyAvicenne HospitalBobignyFrance
  2. 2.Department of OncologyInstitut Gustave RoussyWillejuifFrance
  3. 3.Department of AnatomopathologyAvicenne HospitalBobignyFrance
  4. 4.Department of OncologyInstitut CurieParisFrance
  5. 5.Service d’Oncologie MédicaleHôpital AvicenneBobigny, CedexFrance

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