Letrozole as second-line hormonal treatment for recurrent low-grade endometrial stromal sarcoma: A case report and review of the literature

Nakamura,Kohei; Nakayama,Kentaro; Ishikawa,Masako; Ishikawa,Noriyoshi; Katagiri,Hiroshi; Katagiri,Atsuko; Ishibashi,Tomoka; Sato,Emi; Iida,Kohji; Sultana,Razia; Kyo,Satoru

doi:10.3892/ol.2016.5186

Letrozole as second-line hormonal treatment for recurrent low-grade endometrial stromal sarcoma: A case report and review of the literature

Authors:
- Kohei Nakamura
- Kentaro Nakayama
- Masako Ishikawa
- Noriyoshi Ishikawa
- Hiroshi Katagiri
- Atsuko Katagiri
- Tomoka Ishibashi
- Emi Sato
- Kohji Iida
- Razia Sultana
- Satoru Kyo
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Affiliations: 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
Published online on: September 26, 2016 https://doi.org/10.3892/ol.2016.5186
Pages: 3856-3860
Copyright: © Nakamura et al. This is an open access article distributed under the terms of Creative Commons Attribution License.

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Abstract

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.

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