Blake Erin A, Carter Charelle M, Kashani Banafsheh N, Kodama Michiko, Mabuchi Seiji, Yoshino Kiyoshi, Matsuo Koji
Division of Gynecologic Oncology, Department of Obstetrics and Gynecology, Los Angeles County Medical Center, University of Southern California, USA; Department of Obstetrics and Gynecology, University of Colorado, Denver, CO, USA.
Department of Obstetrics and Gynecology, Georgetown/Washington Hospital Center, Washington, DC, USA.
Eur J Obstet Gynecol Reprod Biol. 2014 Apr;175:1-7. doi: 10.1016/j.ejogrb.2013.12.025. Epub 2013 Dec 25.
Sex-cord stromal tumors (SCSTs) are rare ovarian cancers and their behavior during pregnancy is not well understood. To evaluate the maternal and fetal outcomes of pregnancy complicated by ovarian SCST, a systematic literature search was conducted in PubMed/MEDLINE using entry key words "pregnancy" and each type of ovarian SCST ("sex cord stromal tumor," "granulosa cell tumor," "thecoma," "Sertoli-Leydig cell tumor," or "gynandroblastoma") between 1955 and 2012 that identified 46 cases eligible for the analysis. Clinical characteristics, pregnancy outcome, tumor characteristics, and survival outcomes were evaluated. Serious adverse events were defined as complications related to the SCST that resulted in severe morbidity or mortality for mother, fetus, or both. The most common histology was granulosa cell tumor (22.0%), followed by thecoma (18.6%) and Sertoli-Leydig cell tumor (8.5%). Abdomino-pelvic pain (45.7%), palpable mass (30.4%), and virilization (26.1%) were the three most common symptoms. The majority were stage I (76.1%), tumor size <15cm (64.9%), and underwent unilateral adnexectomy (80.4%). Fetal conservation surgery was seen in 54.3%. Most cases had live births (78.3%) at full term (60.9%). Among cases proceeded expectant delay of delivery (45.7%), most cases resulted in live birth (95.2%) with median expectant interval of 20.7 weeks. Maternal and/or fetal serious adverse events (SAEs) were observed in 41.3% with maternal shock/hemoperitoneum being the most common complication (13.0%). Logistic regression test identified younger age (<30 versus ≥30, 73.3% versus 26.7%, odds ratio [OR] 11.7, 95%CI 1.35-101, p=0.026), large tumor (size ≥15cm versus <15cm, 64.9% versus 35.1%, OR 10.0, 95%CI 1.29-26.2, p=0.004), and advanced-stage (stages II-IV versus I, 76.1% versus 23.9%, OR 5.82, 95%CI 2.05-48.9, p=0.022) as risk factors of increased SAE. Overall survival of patients diagnosed with ovarian SCST during pregnancy was comparable to ovarian SCST not related to pregnancy (5-year rate, stages I and II-IV, 100% and 70.0%, respectively). In conclusion, although the majority of cases resulted in live birth, ovarian SCST-complicated pregnancy falls into the category of high-risk pregnancy. Risk factors for SAE identified in our study will help to guide strategic management of pregnancy complicated by ovarian SCST.
性索间质肿瘤（SCSTs）是一种罕见的卵巢癌，其在孕期的表现尚不明确。为评估合并卵巢性索间质肿瘤的妊娠产妇及胎儿结局，我们在PubMed/MEDLINE数据库中进行了系统的文献检索，使用关键词“妊娠”以及1955年至2012年间各种类型的卵巢性索间质肿瘤（“性索间质肿瘤”、“颗粒细胞瘤”、“卵泡膜瘤”、“支持-间质细胞瘤”或“两性母细胞瘤”）进行检索，共识别出46例符合分析条件的病例。我们评估了临床特征、妊娠结局、肿瘤特征及生存结局。严重不良事件定义为与性索间质肿瘤相关的并发症，导致母亲、胎儿或两者出现严重发病或死亡。最常见的组织学类型是颗粒细胞瘤（22.0%），其次是卵泡膜瘤（18.6%）和支持-间质细胞瘤（8.5%）。腹痛（45.7%）、可触及肿块（30.4%）和男性化（26.1%）是最常见的三种症状。大多数病例为Ⅰ期（占76.1%），肿瘤大小<15cm（占64.9%），并接受了单侧附件切除术（占80.4%）。54.3%的病例进行了保留胎儿的手术。大多数病例足月分娩活婴（占78.3%）（其中60.9%为足月）。在采取期待性延迟分娩的病例中（占45.7%），大多数病例分娩活婴（占95.2%），期待期的中位数为20.7周。41.3%的病例出现了母亲和/或胎儿严重不良事件（SAEs），其中母亲休克/腹腔内出血是最常见的并发症（占13.0%）。逻辑回归分析确定年龄较小（<30岁与≥30岁，分别为73.3%与26.7%，比值比[OR]为11.7，95%置信区间为1.35 - 101，p = 0.026）、肿瘤较大（大小≥15cm与<15cm，分别为64.9%与35.1%，OR为10.0，95%置信区间为1.29 - 26.2，p = 0.004）以及晚期（Ⅱ - Ⅳ期与Ⅰ期，分别为76.1%与23.9%，OR为5.82，95%置信区间为2.05 - 4.89，p = 0.022）是严重不良事件增加的危险因素。孕期诊断为卵巢性索间质肿瘤患者的总体生存率与非妊娠相关的卵巢性索间质肿瘤相当（5年生存率，Ⅰ期和Ⅱ - Ⅳ期分别为100%和70.0%）。总之，尽管大多数病例分娩活婴，但合并卵巢性索间质肿瘤的妊娠仍属于高危妊娠类别。我们研究中确定的严重不良事件危险因素将有助于指导合并卵巢性索间质肿瘤妊娠的策略性管理。
