Kondi-Pafiti A, Bakalianou K, Iavazzo C, Dastamani C, Hasiakos D, Liapis A
Pathology Department, University of Athens, Aretaieion Hospital, Athens, Greece.
Eur J Gynaecol Oncol. 2011;32(4):452-4.
Peutz-Jeghers syndrome is a rare syndrome which is inherited in a dominant manner. It is characterized by hamartomatous polyps of the gastrointestinal tract, hyperpigmented macules of the oral mucosa and an increased risk of developing neoplasms in the gastrointestinal tract, pancreas, breast and genital system. Women with Peutz-Jeughers syndrome often develop an ovarian sex cord tumor and cervical adenocarcinoma of minimal deviation adenoma malignum type. A case of a 58-year-old patient with Peutz-Jeghers syndrome and history of multiple malignancies (thyroid, breast and colon cancer) who presented with metrorrhagia is reported. The dilatation and curettage revealed endometrial adenocarcinoma. The patient underwent total abdominal hysterectomy with bilateral oophorectomy. The histologic examination showed an endometrioid endometrial adenocarcinoma that developed in atypical endometrial hyperplasia. The histologic examination of the right ovary revealed a sex cord tumor with annular tubules, measuring 3 cm. Sex cord tumors with annular tubules in patients with Peutz-Jeghers syndrome are usually small, bilateral tumors of the ovaries which have common characteristics with granulosa cell tumor and Sertoli cell tumor. Hyperestrogenism is a rather common finding with development of estrogen-dependent lesions.
佩-吉综合征是一种罕见的以显性方式遗传的综合征。其特征为胃肠道错构瘤性息肉、口腔黏膜色素沉着斑以及胃肠道、胰腺、乳腺和生殖系统发生肿瘤的风险增加。患有佩-吉综合征的女性常发生卵巢性索肿瘤和微小偏离性腺瘤恶性型宫颈腺癌。本文报告了一例58岁患有佩-吉综合征且有多种恶性肿瘤病史(甲状腺癌、乳腺癌和结肠癌)的患者,该患者出现子宫出血。刮宫术显示为子宫内膜腺癌。患者接受了全腹子宫切除术及双侧卵巢切除术。组织学检查显示为非典型子宫内膜增生发展而来的子宫内膜样腺癌。右侧卵巢的组织学检查发现一个环状小管性索肿瘤,大小为3厘米。佩-吉综合征患者的环状小管性索肿瘤通常为双侧小卵巢肿瘤,与颗粒细胞瘤和支持细胞瘤具有共同特征。高雌激素血症是雌激素依赖性病变发生时相当常见的表现。