Complete remission of advanced low-grade endometrial stromal sarcoma after aromatase inhibitor therapy: a case report (original) (raw)
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Archives of Gynecology and Obstetrics, 2009
Background Approximately 50% of patients with lowgrade endometrial stromal sarcoma (ESS) develop recurrent disease including mainly metastases of pelvis and lung. A solitary pulmonary metastasis of this disease is a rare phenomenon. Case After initial surgical treatment in 1990, a 28-year-old female developed solitary metastases of the lung in 1998 and 1999, which were surgically removed both times. The patient was then treated with goserelin for 2 years followed by anastrozole to date. She has since been disease-free. Conclusions This case report emphasizes the importance of long-term follow-up and initial surgical treatment of low-grade ESS and suggests systemic hormonal therapy in case of recurrence.
Geburtshilfe und Frauenheilkunde, 2008
Background Approximately 50% of patients with lowgrade endometrial stromal sarcoma (ESS) develop recurrent disease including mainly metastases of pelvis and lung. A solitary pulmonary metastasis of this disease is a rare phenomenon. Case After initial surgical treatment in 1990, a 28-year-old female developed solitary metastases of the lung in 1998 and 1999, which were surgically removed both times. The patient was then treated with goserelin for 2 years followed by anastrozole to date. She has since been disease-free. Conclusions This case report emphasizes the importance of long-term follow-up and initial surgical treatment of low-grade ESS and suggests systemic hormonal therapy in case of recurrence.
Clinical Sarcoma Research, 2015
Background: Hormonal manipulation is sometimes recommended in the treatment of metastatic endometrial stromal sarcoma, but there are few data assessing the efficacy of endocrine therapies in this subtype of uterine sarcomas. Methods: We performed a retrospective electronic medical record review of patients with metastatic ESS treated with a hormonal agent at Royal Marsden Hospital between 1999 and 2011. We assessed progression-free survival (PFS), objective response and toxicity profile among patients with measurable disease. Results: Thirteen patients with metastatic ESS were treated with hormonal therapies. Hormone receptor status (estrogen and progesterone receptors) was assessed in 9 out of 13 patients and in all of them it was moderately to strongly positive. Aromatase inhibitors (AIs) were prescribed as first endocrine line in 11/13 patients and progestins in the remainder, while in 2 nd line treatment AIs were prescribed in 7/10 patients, followed by progestins and GnRH analogues. Median PFS for 1 st line was 4.0 years (95% CI: 2.4-5.5 years) with 5-year progression-free rate of 30.8% (95% CI: 5.7-55.9%), both of which reflect the indolent natural history of ESS. Best objective response was partial response (PR) in 6/13 patients (46.2%; 95% CI: 19.2-74.9) and clinical benefit rate (defined as complete response + PR + stable disease ≥6 months) was 92.4% (95% CI: 64.0-99.8%; 12/13 patients). Median PFS for 2 nd line was 3.0 years (95% CI: 2.0-4.1 years) with 2-year progression-free rate of 88.9% (95% CI: 68.3-100.0). Conclusions: In this cohort of metastatic ESS patients, 1st line endocrine treatment achieved objective response in 46.2% of them and clinical benefit in 92.4%. Tamoxifen and hormone replacement therapy should not be prescribed in patients with ESS due to their detrimental effects. Until more solid data are available, a reasonable recommendation would be that 1 st line treatment with an endocrine treatment, preferably with an AI. Moreover, in view of the positive outcomes of our patients that received 2 nd /3 rd line endocrine treatments, all available hormonal options should be used in sequence in the management of ESS.
High-Grade Endometrial Stromal Sarcoma after Tamoxifen Therapy for Breast Cancer
Gynecologic and Obstetric Investigation, 2005
A case of high-grade endometrial stromal sarcoma, confi ned into an intrauterine polypoid growth, in a woman with a history of breast cancer who was treated with adjuvant tamoxifen. Based on the fi ndings, a high-grade endometrial stromal sarcoma was diagnosed. The patient underwent a total hysterectomy and bilateral salpingo-oophorectomy with multiple omental biopsies. Pathological examination on multiple uterine sections showed the absence of residual tumor cells in the uterus. The endometrium showed patterns of glandular cystic hyperplasia. After 14 months of follow-up, the patient is well and free of disease. In deciding if tamoxifen therapy is warranted, all potentially life-threatening adverse events associated with tamoxifen should be considered, including endometrial adenocarcinoma or uterine sarcoma.
Journal of Medical Case Reports, 2017
Background: Chemotherapy is considered the most appropriate treatment for metastatic uterine sarcoma, despite its limited efficacy. No other treatment has been conclusively proved to be a real alternative, but some reports suggest that anti-hormonal therapy could be active in a small subset of patients. We report the case of a patient with metastatic uterine carcinosarcoma with positive hormonal receptors and a complete pathological response. Case presentation: A 54-year-old white woman presented to our emergency room with hypovolemic shock and serious vaginal bleeding. After stabilization, she was diagnosed as having a locally advanced uterine carcinosarcoma with lymph nodes and bone metastatic disease. In order to control the bleeding, palliative radiotherapy was administered. Based on the fact that positive hormone receptors were found in the biopsy, non-steroidal aromatase inhibitor therapy with letrozole was started. In the following weeks, her general status improved and restaging imaging tests demonstrated a partial response of the primary tumor. Ten months after initiating aromatase inhibitor therapy, she underwent a radical hysterectomy and the pathological report showed a complete response. After completing 5 years of treatment, aromatase inhibitor therapy was stopped. She currently continues free of disease, without further therapy, and maintains a normal and active life. Conclusions: This case shows that patients with uterine carcinosarcoma and positive hormone receptors may benefit from aromatase inhibitor therapy. A multidisciplinary strategy that includes local therapies such as radiation and/ or surgery should be considered the mainstay of treatment. Systemic therapies such as hormone inhibitors should be taken into consideration and deserve further clinical research in the era of precision medicine.
Aromatase inhibitors in recurrent ovarian endometriomas: report of five cases with literature review
Fertility and Sterility, 2011
Objective: To determine the role of the aromatase inhibitor letrozole in the treatment of recurrent ovarian endometrioma cases. Design: Nonrandomized proof of concept study. Settings: Outpatient tertiary-care center. Patient(s): Five premenopausal patients with documented ovarian endometriomas and chronic pelvic pain, all of whome were previously treated with surgery and medicine with unsatisfactory results. Intervention(s): Ovarian endometriomas were diagnosed by biopsy after laparoscopic ovarian cystectomy and subsequently treated with hormomes. After a 6-month washout of endometriosis hormone therapies, women took letrozole (2.5 mg), one tablet of 0.15 mg of desogestrel, and 0.03 mg of ethinyl estradiol, calcium (1,200 mg), and vitamin D 3 (800 IU) daily for 6 months. Main Outcome Measure(s): Size of endometriomas (monitored by ultrasound), assessment of pelvic pain (by visual analog scale), and bone density (DEXA scan). Result(s): Disappearance of ovarian endometrioma and reduction in pelvic pain in all cases at the end of 6 months. The size of ovarian endometriomas was reduced after 3 months. Pain scores decreased only after 1 month of treatment and continued decreasing in each treatment month. Overall, no significant change in bone density was detected. Conclusion(s): Letrozole given with combined pills achieved complete regression of recurrent endometriotic cysts and pain relief in all cases. (Fertil Steril Ò 2011;95:291.e15-e18. Ó2011 by American Society for Reproductive Medicine.
Aromatase Inhibitors and Endometrial Adenocarcinoma: The Role of Hormone Receptor Assessment
Journal of Gynecology and Womens Health, 2018
Although adenocarcinoma of the endometrium (EC) is a hormone dependent neoplasm, no hormone therapy has ever been registered and there are no clear guidelines for the determination of steroid hormone receptors in the tumor tissue. Aromatase inhibitors (AIs) have been hypothesized as an opportunity for treatment. This article briefly reviews the current evidence for the role of AIs and the potential prognostic/ predictive role of hormone receptors in the treatment of EC.
British journal of cancer, 2007
The objective of this study is to assess the therapeutic importance of surgical castration, adjuvant hormonal treatment and lymphadenectomy in endometrial stromal sarcoma (ESS). A retrospective and multicentric search was performed. Clinicopathologic data were retrieved from cases that were confirmed to be ESS after central pathology review. The protocol was approved by the Ethical Committee. ESS was confirmed histopathologically in 34 women, but follow-up data were available in only 31 women. Surgical treatment (n=31) included hysterectomy with or without bilateral salpingo-oophorectomy (BSO) in 23 out of 31 (74%) and 8 out of 31 (26%) cases, respectively. Debulking surgery was performed in 6 out of 31 cases (19%). Stage distribution was as follows: 22 stage I, 4 stage III and 5 stage IV. Women with stage I disease recurred in 4 out of 22 (18%) cases. Among stage I women undergoing hormonal treatment with or without BSO, 3 out of 15 (20%) and 1 out of 7 (14%) relapsed, respectively...
Aromatase and comparative response to its inhibitors in two types of endometrial cancer
The Journal of Steroid Biochemistry and Molecular Biology, 2005
Aromatase activity (AA) was evaluated totally in 80 tumors collected from primary endometrial cancer (EC) patients. All patients were divided into cases belonging to the types I or II of EC (respectively, 50 and 30 observations). Samples of malignant endometrium from type II demonstrated inclination to the higher AA in comparison with type I samples; the difference reached level of statistical significance in non-smoking patients (p = 0.02). Although no positive correlation was revealed between AA in EC tissue and percentage of cells with DNA damage in normal endometrium from the same patients, the rate of DNA damage (percent of comets, comet's tail average length, etc.) was higher in intact endometrium collected from patients with type II of the disease. In 19 tumor samples, CYP19 gene expression was evaluated by RT-PCR and level of mRNA signal demonstrated positive correlation with AA (R s = +0.63, p = 0.05) in the whole this material. Of note, though, CYP19 mRNA expression was not revealed in six cases, and all of them belonged to the type I of disease. Finally, in 23 EC patients (15 with type I and 8 with type II of the disease) effects of 2 weeks treatment with letrozole (10 pts) and exemestane (13 pts) were evaluated in neoadjuvant setting. Although diminishing of endometrial M-echo signal and the increases in FSH and LH concentration after treatment were more pronounced in type I patients, decrease in tumor PR content (p = 0.04) was more revealing in patients with type II of EC; besides, the decreases in AA in tumor tissue by the end of treatment were noted predominantly in patients with lower body weight (BMI <27.5). Thus, although type II of EC is frequently considered as hormone-independent, increased ability of this type of the tumor to estrogen biosynthesis (at CYP19 gene and protein level) may lead to the reconsideration of such conclusion and warrants further investigation. The search of possible ethnic differences in AA and in the biologic response to aromatase inhibitors in EC can be of importance too.