Leuprolide和来曲唑联合治疗复发性卵巢成人颗粒细胞瘤延长无病生存期:病例报告

IF 1.1 Q4 OBSTETRICS & GYNECOLOGY
G. Garuti, P. Sagrada, Susanna Delfrati, Lorenzo Sogaro, M. Soligo
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引用次数: 0

摘要

由于缺乏有效的治疗,复发性卵巢颗粒细胞瘤(GCT)对医生来说是一个挑战。目前的策略并没有改善80%的复发性疾病死亡率。GCTs合成雌激素并表达促卵泡激素、促性腺激素释放激素以及雌激素和孕激素受体。FOXL2-C134W突变在所有GCT中都有,其对激素相关细胞凋亡的下调似乎是诱导肿瘤表型的原因。基于这些假设,激素抗雌激素疗法已被提议用于复发性GCT。2004年,一名患有GCT的32岁女性首次接受手术治疗,并被诊断为IA病。随后在2006年和2007年诊断出两次盆腔复发,患者接受了手术和化疗来治疗这两次复发。总的来说,她随后接受了五次手术干预和两次化疗。2013年诊断出第三例阴道套以上盆腔复发。基于患者拒绝接受进一步手术,我们提出了一种由GnRH类似物亮丙瑞林和芳香化酶抑制剂来曲唑联合组成的抗雌激素疗法。从治疗开始3个月后获得完全缓解。随后,我们发现,经过9年的治疗,无病生存率得以维持。尽管最近的报告表明激素治疗复发性GCT的有效性较差,但该病例的成功表明,复发性GCT的一部分患者对抗雌激素药物保持着高度反应的肿瘤表型。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Prolonged Disease-Free Survival in a Relapsed Adult Granulosa Cell Tumor of the Ovary Treated by Combined Leuprolide and Letrozole: Case Report
Relapsing ovarian granulosa-cell tumor (GCT) is a challenge for physicians due to the lack of effective therapy. Current strategies did not improve the 80% death rate of recurrent disease. GCTs synthesize estrogens and express follicle-stimulating hormone, gonadotropin-releasing hormone, and estrogen and progesterone receptors. The FOXL2-C134W mutation is shared in all GCTs, and its downregulation of hormone-related apoptosis appears causal in induction of tumor phenotype. On these assumptions, hormone anti-estrogenic therapies have been proposed for recurrent GCTs. A 32-year-old woman suffering from GCT was first treated by surgery in 2004 and staged as IA disease. Two subsequent pelvic relapses were diagnosed in 2006 and 2007, and the patient underwent surgery and chemotherapy to treat both recurrences. Overall, she underwent five subsequent surgical interventions and two chemotherapy instances. A third single pelvic relapse above the vaginal cuff was diagnosed in 2013. Based on the patient’s refusal to undergo further surgery we proposed an anti-estrogen therapy consisting of combined GnRH analogue leuprolide and the aromatase inhibitor letrozole. Complete remission was obtained after 3 months from the start of therapy. Subsequently, we found that disease-free survival was maintained over 9 years of treatment. Although recent reports indicate poor effectiveness of hormone therapy to treat recurrent GCTs, the success of this case indicates that a subset of patients with recurrent GCT maintain a tumor phenotype highly responsive to anti-estrogen drugs.
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