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

doi:10.3892/ol.2015.3674

Cited by 24 publications

(21 citation statements)

References 24 publications

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“…Therefore, hormone replacement therapy for menopausal syndrome is contraindicated, and progestins (megestrol and medroxyprogesterone acetate) or aromatase inhibitors are the therapeutic choice in managing women with LG-ESS, acting as postoperative adjuvant therapy for residual or recurrent diseases. [ 15 ] A Phase II study showed that single-agent mifepristone (RU-486) in managing LG-ESS could result in a stable disease rate of 50%. [ 16 ]…”

Section: Discussionmentioning

confidence: 99%

Endometrial stromal sarcoma in combination with mixed type endometrial carcinomas

Xiu

Wang²,

Lv³

et al. 2017

Medicine

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Rationale:Endometrial stromal sarcoma (ESS) is rare, representing only approximately 0.2% of all uterine malignancies. Mixed type endometrial carcinomas (MT-ECs) are rare tumors with both type I and II features, and are difficult to diagnose. Cases of ESS and MT-ECs coexisting in the same patient are extremely rare. This study aimed to describe a case of ESS in combination with MT-ECs in a 47-year-old premenopausal woman.Patient concerns:A woman presented to the hospital complaining of occasional abdominal pain and had high tumor markers: cancer antigen (CA) 19–9 (263.6 U/mL) and CA 125 (428.0 U/mL). Transvaginal ultrasound examination revealed a complex mass (12.3 × 9.1 × 6.3 cm) with solid and cystic components on the right rear wall of the uterus. Abdominopelvic computed tomography images showed a pelvic cystic-solid mixed mass. The patient underwent an exploratory midline laparotomy. The mass was hypothesized to be malignant on the uterine posterior wall. Tumor deposits were found on bilateral parametrium. On peritoneal implantation, multiple metastases were seen on the serosal surface of the bowel and greater omentum. A frozen section revealed a spindle cell sarcoma.Diagnoses:Pathological reports following surgery revealed concurrent ESS and MT-ECs.Interventions:The patient underwent a total abdominal hysterectomy, bilateral salpingo-oophorectomy, total omentectomy, and macroscopic clearance of the tumor. Adjuvant chemotherapy was given.Outcomes:The patient was still alive when this report was written.Lessons:Considering the rarity of ESS in combination with MT-ECs, this study presented an overview of the literature and discussed a number of histological and clinical issues. Nevertheless, etiology and pathogenesis of these tumors need further investigation.

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Section: Discussionmentioning

confidence: 99%

Endometrial stromal sarcoma in combination with mixed type endometrial carcinomas

Xiu

Wang²,

Lv³

et al. 2017

Medicine

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“…A response rate of 88% to progestin therapy was reported by Chu et al in eight women with recurrent low-grade endometrial stromal sarcoma 11. Aromatase inhibitors also represent an attractive option with a response rate of 77.4% in a review of data from 30 patients 51. Optimal agents and dosage are yet to be established.…”

Section: Discussionmentioning

confidence: 98%

Ovarian preservation for low-grade endometrial stromal sarcoma: a systematic review of the literature and meta-analysis

Nasioudis

Kolovos³

et al. 2019

Int J Gynecol Cancer

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ObjectiveTo evaluate the effect of ovarian preservation on oncologic outcomes for women with low-grade endometrial stromal sarcoma of the uterus.MethodsA systematic search of the Medline, Embase, Cohrane, and Web of Science databases was performed based on the Preferred Reporting Items for Systematic Reviews and Meta-analyses guidelines. Studies including patients with low-grade endometrial stromal sarcoma who had hysterectomy were identified. Data on tumor recurrence and death rate were pooled using a random effects model.ResultsA total of 17 studies met the inclusion criteria and reported on 786 patients. Based on available information, ovarian preservation was noted in 190 patients while 501 had bilateral salpingo-oophorectomy. A significantly increased tumor recurrence rate was observed in the ovarian preservation group (89/190, 46.8%) compared with the bilateral salpingo-oophorectomy group (121/501, 24.2%) (OR 2.70, 95% CI 1.39 to 5.28). Based on data from 162 patients, no difference in death rate was noted between the ovarian preservation (2/34, 5.9%) and bilateral salpingo-oophorectomy (9/128, 7%) groups (OR 0.80, 95% CI 0.18 to 3.47).ConclusionsApproximately one-quarter of patients with low-grade endometrial stromal sarcoma were managed with ovarian preservation. These women experienced a higher recurrence rate. Hormone exposure may be responsible for this elevated risk. Given the apparent high salvage rate, however, ovarian preservation may be an option only in a well-informed patient population.

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“…In unresectable disease, endocrine therapy is thought to be the treatment of choice since ESS is considered to be relatively radio-and chemo-therapy resistant. Under hormonal therapy, partial response (PR) or disease stabilization (SD) of ESS le- [7][8][9][10][11][12][13][14][15][16][17][18][19][20][21][22][23][24]. AIs were used as first-line or second-line (or later-line) treatment, salvage after prior chemotherapy or adjuvant therapy after complete resection of the metastatic lesions.…”

Section: Discussionmentioning

confidence: 99%

Aromatase inhibitor therapy for endometrial stromal sarcoma — two-centre experience

et al. 2018

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Objectives: Endocrine therapy is the recommended systemic treatment for steroid receptor positive endometrial stromal sarcoma (ESS). There is no current consensus on the optimal hormonal therapy for ESS. The literature offers several reports on advanced/recurrent/metastatic ESS patients treated with progestins, whereas data on the efficacy of aromatase inhibitors are scarce. Material and methods: We retrospectively identified cases treated for ESS with aromatase inhibitors at our institutions. There were five patients with advanced or unresectable recurrent estrogen, progesterone and androgen receptor-positive ESS, treated with aromatase inhibitors: letrozole or anastrozole (at a daily dose of 2.5 mg and 1 mg, respectively), as first-line endocrine therapy in all but one case treated following progression with megestrol acetate. Results: Disease stabilization was achieved in four cases (80%), including two with long-term progression-free survival for up to 10 years attained under letrozole treatment, and one case after prior progestin treatment. During therapy, no substantial toxicity was observed. Conclusions: Aromatase inhibitors as first-or second-line endocrine treatment achieve disease control in most steroid receptor positive ESS. Our series of cases is evidence of aromatase inhibitors efficacy as long-term endocrine treatment option for ESS patients.

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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

Abstract: Abstract. Endometrial stromal sarcoma (ESS) occurs rarely and accounts for only 0.2% of all uterine malignancies.

Cited by 24 publications

References 24 publications

Endometrial stromal sarcoma in combination with mixed type endometrial carcinomas

Endometrial stromal sarcoma in combination with mixed type endometrial carcinomas

Ovarian preservation for low-grade endometrial stromal sarcoma: a systematic review of the literature and meta-analysis

Aromatase inhibitor therapy for endometrial stromal sarcoma — two-centre experience

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