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摘要: 卵巢硬化性间质瘤(ovarian sclerosing stromal tumor, OSST)是一种罕见的良性卵巢性索间质肿瘤，目前文献报道的病例总数约为100例。OSST主要发病群体为20~30岁育龄期女性，临床表现为腹痛、附件包块，肿瘤可具有内分泌功能，易导致月经紊乱、不孕，少数患者出现多毛痤疮、声音低沉等男性化表现。该病在青春期前女童中极为罕见，半数OSST患儿为体检时发现盆腔肿物，也有因性激素升高引起临床表现而就诊。其通常为单侧卵巢发病，目前仅有3例报道发生于双侧卵巢。对于双侧OSST的年轻女性患者，可行双侧卵巢肿物切除术或至少保留一侧正常卵巢组织手术，最大程度保留患者生育力。该病预后良好且术后随访均未见复发及转移。本文报道1例罕见的青春期前女童双侧OSST病例，并通过相关文献复习对该疾病的临床特征、鉴别诊断、治疗及预后进行讨论，以提高临床医生对该疾病的认识。Abstract: Ovarian sclerosing stromal tumor(OSST)is a benign ovarian sex cord stromal tumor. There are about 100 cases reported in literature. OSST usually occurs in women of childbearing age from 20 to 30 years old. The clinical manifestations are abdominal pain and adnexal mass. The tumor may have endocrine function, causing menstrual disorder and infertility, and a few patients even appear masculine. OSST is rare in prepubertal girls. Half of the patients go to see a doctor due to the accidental discovery of pelvic tumors during physical examination or the clinical manifestations caused by the increase of sex hormones. It usually occurs in one side of the ovary. Only 3 cases reported tumors in bilateral ovaries. For young women with bilateral OSST, bilateral ovarian tumor resection or operation with normal ovarian tissue on one side can be performed to preserve the fertility of the patients. In this paper, we report a rare case of bilateral OSST of the ovaries in a premenstrual girl and review the literature.作者贡献：刘阳负责查阅文献、撰写论文；周慧梅负责收集数据、修订论文；曹冬焱、杨佳欣负责论文构思、写作指导；向阳、郎景和负责论文修订及审校。利益冲突：所有作者均声明不存在利益冲突
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图 1 患儿术前腹盆影像学检查
A. CT检查示盆腔内11.3 cm×9.0 cm软组织团块，内见多发形态不规则钙化及局限性低密度区；B、C. 增强MRI检查示盆腔类圆形混杂信号占位，大小约为9.0 cm×7.6 cm×7.4 cm，DWI稍高信号，其内多发片状长T1、长T2坏死区，增强不强化，另可见中央长条状长T1、短T2信号影，增强不强化，余病变实性成分可见中度强化
图 3 术后病理诊断符合双侧卵巢硬化性间质瘤
A. HE染色(×100)；B. HE染色(×200)；C. Calretinin染色(部分+)；D. α-inhibin染色(+)
表 1 7例青春期前OSST患儿临床表现
第一作者 发表年份 年龄 表现 性激素 肿瘤标志物 肿瘤侧别及大小(cm) 手术方式 随访情况 Yen[8] 2014 9岁 男性化 雄激素升高 正常 左侧15.0×8.5×6.0 左附件切除 随访6个月：术后GnRH-a治疗，性激素恢复正常，男性化症状持续 Chang[5] 2009 12岁 无 未提及 正常 右侧13.0; 左侧小结节 右附件+左卵巢楔形切除+大网膜切除 未提及 Chang[3] 2006 11岁 无 未提及 正常 右侧17.0×12.0×8.0; 左侧8.5×6.0×5.0 双附件切除 时间不详：术后长期激素补充治疗 Duke[4] 2008 7个月 阴道出血 孕酮升高 正常 右侧5.5×4.5×2.5 右卵巢切除 时间不详：术后阴道出血停止 Park[7] 2011 11岁 男性化 雄激素升高 未提及 左侧9.0×7.5×6.0 左附件切除 随访6个月：术后性激素恢复正常；3个月初潮，6个月乳房发育，男性化症状缓解 Fefferman[2] 2003 10岁 无 未提及 正常 左侧13.0×10.0×8.0 左附件切除+右卵巢活检 未提及 He[6] 2010 4岁 乳房发育 正常 正常 右侧4.4×3.6×3.4 肿物切除 随访4个月 OSST: 卵巢硬化性间质瘤 
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[1] Chalvardjian A, Scully RE. Sclerosing stromal tumors of the ovary[J]. Cancer, 1973, 31: 664-670. doi: 10.1002/1097-0142(197303)31:3<664::AID-CNCR2820310327>3.0.CO;2-F [2] Fefferman NR, Pinkney LP, Rivera R, et al. Sclerosing stromal tumor of the ovary in a premenarchal female[J]. Pediatr Radiol, 2003, 33: 56-58. doi: 10.1007/s00247-002-0694-7 [3] Chang W, Oiseth SJ, Orentlicher R, et al. Bilateral sclerosing stromal tumor of the ovaries in a premenarchal girl[J]. Gynecol Oncol, 2006, 101: 342-345. doi: 10.1016/j.ygyno.2005.10.041 [4] Duke DS, Yoo EY, Newton C, et al. A rare cause of vaginal bleeding in a 7-month-old female infant[J]. J Pediatr Surg, 2008, 43: e1-e4. [5] Chang YW, Hong SS, Jeen YM, et al. Bilateral sclerosing stromal tumor of the ovary in a premenarchal girl[J]. Pediatr Radiol, 2009, 39: 731-734. doi: 10.1007/s00247-009-1190-0 [6] He Y, Yang KX, Jiang W, et al. Sclerosing stromal tumor of the ovary in a 4-year-old girl with characteristics of an ovarian signet-ring stromal tumor[J]. Pathol Res Pract, 2010, 206: 338-341. doi: 10.1016/j.prp.2009.06.005 [7] Park SM, Kim YN, Woo YJ, et al. A sclerosing stromal tumor of the ovary with masculinization in a premenarchal girl[J]. Korean J Pediatr, 2011, 54: 224-227. doi: 10.3345/kjp.2011.54.5.224 [8] Yen E, Deen M, Marshall I. Youngest Reported Patient Presenting with an Androgen Producing Sclerosing Stromal Ovarian Tumor[J]. J Pediatr Adolesc Gynecol, 2014, 27: e121-e124. doi: 10.1016/j.jpag.2013.09.007 [9] Naidu A, Chung B, Simon M, et al. Bilateral Sclerosing Stromal Ovarian Tumor in an Adolescent[J]. Case Rep Radiol, 2015, 2015: 271394. [10] Torricelli P, Caruso Lombardi A, Boselli F, et al. Sclerosing stromal tumor of the ovary: US, CT, and MRI findings[J]. Abdom Imaging, 2002, 27: 588-591. doi: 10.1007/s00261-001-0096-6 [11] Gordon A, Lipton D, Woodruff JD. Dysgerminoma: a review of 158 cases from the Emil Novak Ovarian Tumor Registry[J]. Obstet Gynecol, 1981, 58: 497-504. [12] Pressley RH, Muntz HG, Falkenberry S, et al. Serum lactic dehydrogenase as a tumor marker in dysgerminoma[J]. Gynecol Oncol, 1992, 44: 281-283. doi: 10.1016/0090-8258(92)90058-Q [13] Zhang HY, Zhu JE, Wen H, et al. Clinicopathologic features of ovarian Sertoli-Leydig cell tumors[J]. Int J Clin Exp Pathol, 2014, 7: 6956-6964. [14] 王子毅, 孙爱军, 郎景和. 卵巢类固醇细胞瘤14例临床分析[J]. 生殖医学杂志, 2010, 19: 6-12. https://www.cnki.com.cn/Article/CJFDTOTAL-SZYX201001007.htm -
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