Nakamura Kohei, Nakayama Kentaro, Ishikawa Masako, Ishikawa Noriyoshi, Katagiri Hiroshi, Katagiri Atsuko, Ishibashi Tomoka, Sato Emi, Iida Kohji, Sultana Razia, Kyo Satoru
Department of Obstetrics and Gynecology, Shimane University School of Medicine, Izumo, Shimane 6938501, Japan.
Department of Organ Pathology, Shimane University School of Medicine, Izumo, Shimane 6938501, Japan.
Oncol Lett. 2016 Nov;12(5):3856-3860. doi: 10.3892/ol.2016.5186. Epub 2016 Sep 26.
Low-grade endometrial stromal sarcoma (LGESS) is a rare malignancy. The tumor is reportedly responsive to hormonal therapy, most commonly with medroxyprogesterone acetate (MPA), but the effectiveness of aromatase inhibitors for recurrent LGESS remains unclear. The present study reports a case of stage IC LGESS presenting with abnormal uterine bleeding, and also provides a review of the literature. Following a total abdominal hysterectomy and bilateral salpingo-oophorectomy, MPA therapy was initiated; treatment was successful, but discontinued 19 months later due to disruptive side effects. A further 2 months later, the patient presented with recurrent disease and received chemotherapy. MPA treatment was restarted with a partial response. A second recurrence, 4 years later, presented with lung and para-aortic lymph node metastases. The patient responded to treatment with the aromatase inhibitor letrozole. The patient has since exhibited stable disease and remained free of symptoms for 7 years. This case suggests that aromatase-inhibitor treatment may be effective for recurrent LGESS as a second-line treatment.
低级别子宫内膜间质肉瘤（LGESS）是一种罕见的恶性肿瘤。据报道，该肿瘤对激素治疗有反应，最常用的是醋酸甲羟孕酮（MPA），但芳香化酶抑制剂对复发性LGESS的有效性仍不明确。本研究报告了一例表现为子宫异常出血的IC期LGESS病例，并对相关文献进行了综述。在进行全腹子宫切除术和双侧输卵管卵巢切除术后，开始使用MPA治疗；治疗取得成功，但19个月后因严重的副作用而停药。又过了2个月，患者出现疾病复发并接受了化疗。重新开始使用MPA治疗后有部分缓解。4年后第二次复发，出现肺部和主动脉旁淋巴结转移。患者对芳香化酶抑制剂来曲唑治疗有反应。此后，患者病情稳定，7年来一直无症状。该病例表明，芳香化酶抑制剂治疗作为二线治疗可能对复发性LGESS有效。
