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卵巢原发性骨肉瘤:一例报告并文献复习

Primary osteosarcoma of the ovary: a case report and literature review.

作者信息

Fadare Oluwole, Bossuyt Veerle, Martel Maritza, Parkash Vinita

机构信息

Department of Pathology, Yale-New Haven Hospital and Yale University School of Medicine, New Haven, Connecticut, USA.

出版信息

Int J Gynecol Pathol. 2007 Jan;26(1):21-5. doi: 10.1097/01.pgp.0000225840.36750.a2.

DOI:10.1097/01.pgp.0000225840.36750.a2
PMID:17197892
Abstract

Pure osteosarcomas, in which this tumor is not the mesenchymal component of a biphasic tumor, are only rarely encountered in the ovary. Herein, we report a case of a pure ovarian osteoarcoma and review all previously reported cases of ovarian involvement by osteosarcoma in the English literature. The latter includes (with the present case) 6 cases of pure osteosarcoma (Group A), 5 cases of osteosarcoma arising from a teratoma (Group B), and 2 cases of osteosarcomas of osseous origin (humerus and maxilla) metastatic to the ovary (Group C). The average ages of the Groups A, B, and C patients were 52.6, 52, and 37.5 years, respectively. The average tumor sizes in these groups were 13.46, 13.82, and 16 cm, respectively. There was no statististically significant difference between the Group A and B patients with respect to tumor size (P = 0.94) or age (P = 0.96) (t test). Eight of the 11 patients in groups A and B were FIGO stage 3 or 4 at presentation. Five (83%) of the 6 group A patients and 2 (40%) of the 5 group B patients died of their disease an average of 4.8 and 3.5 months after initial evaluations respectively. However, the 2 patients who presented with stage 1 disease (and with follow-up information) have shown no evidence of tumor recurrence at an average of 26 months' follow-up. It is concluded that ovarian osteosarcomas are rare malignancies that are frequently associated with teratomas but may be seen in pure form. Unlike osseous osteosarcoma whose peak age of incidence is in the second decade, ovarian osteosarcomas occur at an older age group. Most patients have advanced stage disease at presentation. Ovarian osteosarcomas have a high (>60%) case fatality rate; analysis of the reported cases suggests that the latter is attributable to the advanced stage at which most patients present, as there are no reported cases of patients with stage 1 disease whose diseases have recurred after surgical resection or who have died of their disease.

摘要

纯骨肉瘤(即该肿瘤并非双相肿瘤的间充质成分)在卵巢中极为罕见。在此,我们报告一例纯卵巢骨肉瘤病例,并回顾英文文献中所有先前报道的骨肉瘤累及卵巢的病例。后者(包括本病例)有6例纯骨肉瘤(A组)、5例源自畸胎瘤的骨肉瘤(B组)以及2例骨源性(肱骨和上颌骨)骨肉瘤转移至卵巢的病例(C组)。A、B、C三组患者的平均年龄分别为52.6岁、52岁和37.5岁。这些组中的肿瘤平均大小分别为13.46厘米、13.82厘米和16厘米。A组和B组患者在肿瘤大小(P = 0.94)或年龄(P = 0.96)方面(t检验)无统计学显著差异。A组和B组的11例患者中有8例在初诊时为国际妇产科联盟(FIGO)3期或4期。A组6例患者中有5例(83%)、B组5例患者中有2例(40%)分别在初次评估后平均4.8个月和3.5个月死于疾病。然而,2例表现为1期疾病(且有随访信息)的患者在平均26个月的随访中未显示肿瘤复发迹象。结论是卵巢骨肉瘤是罕见的恶性肿瘤,常与畸胎瘤相关,但也可能以纯形式出现。与发病高峰年龄在第二个十年的骨骨肉瘤不同,卵巢骨肉瘤发生于年龄较大的人群。大多数患者在初诊时处于疾病晚期。卵巢骨肉瘤的病死率很高(>60%);对报道病例的分析表明,后者归因于大多数患者就诊时处于晚期,因为没有报道1期疾病患者在手术切除后疾病复发或死于疾病的病例。

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