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成功妊娠案例报告及文献复习:16 年后无复发,单独接受活检和化疗的宫颈胚胎性横纹肌肉瘤女性

Successful pregnancy in a female with embryonal rhabdomyosarcoma of the cervix who received biopsy and chemotherapy alone without recurrence after 16 years: a case report and literature review.

机构信息

Department of Obstetrics and Gynecology, West China Second University Hospital, Sichuan University, Number 20, 3rd section, South Renmin Road, Chengdu, 610041, Sichuan, China.

Key Laboratory of Birth Defects and Related Diseases of Women and Children (Sichuan University), Ministry of Education, Number 17, 3rd section, South Renmin Road, Chengdu, 610041, Sichuan, China.

出版信息

BMC Womens Health. 2023 Sep 1;23(1):466. doi: 10.1186/s12905-023-02623-6.

DOI:10.1186/s12905-023-02623-6
PMID:37658404
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC10474710/
Abstract

BACKGROUND

Embryonal rhabdomyosarcoma (ERMS) of the uterine cervix is rare, but the population affected is mostly underage females. The scope of surgery has now evolved from extensive to limited, and organ-preserving surgery combined with chemotherapy is recommended to preserve the patient's fertility. However, reports of birth outcomes are rare.

CASE

A minor woman with cervical ERMS who underwent only an outpatient biopsy of the lesion had no residual lesion on subsequent multipoint cervical biopsy and refused radical surgery or cervical conization, after which the patient received a nonclassical regimen of chemotherapy. The patient stopped the chemotherapy on her own, but the patient conceived spontaneously 16 years later with a good pregnancy outcome and no recurrence.

CONCLUSIONS

This case suggests that preservation of reproductive function is often feasible in immature women with cervical ERMS, and the prognosis is usually good as long as the primary tumour can be surgically removed and the lesion is free of residual disease. We also look forward to reports of subsequent growth and pregnancy outcomes in other children with reproductive tract RMS. In cervical ERMS, accurate evaluation of the disease and development of an individualized treatment plan are crucial, and the protection of reproductive function and psychological well-being deserves special attention.

摘要

背景

宫颈胚胎性横纹肌肉瘤(ERMS)较为罕见,但受影响的人群大多为未成年女性。目前手术范围已从广泛转为局限,建议采用保留器官的手术联合化疗,以保留患者的生育能力。然而,关于生育结局的报道较为少见。

病例

一位未成年女性,宫颈 ERMS 患者,仅行病变部位的门诊活检,后续多点宫颈活检未见残留病灶,且拒绝根治性手术或宫颈锥切术,此后该患者接受了非典型的化疗方案。患者自行停止化疗,但 16 年后自然妊娠,妊娠结局良好,无复发。

结论

本病例提示,对于宫颈 ERMS 的未成年女性,保留生育功能通常是可行的,只要能手术切除原发肿瘤且无残留病灶,预后通常较好。我们也期待其他生殖道 RMS 患儿的后续生长和妊娠结局的报道。在宫颈 ERMS 中,准确评估疾病并制定个体化治疗方案至关重要,保护生殖功能和心理健康值得特别关注。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/7a7f/10474710/4c2cba71f595/12905_2023_2623_Fig1_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/7a7f/10474710/4c2cba71f595/12905_2023_2623_Fig1_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/7a7f/10474710/4c2cba71f595/12905_2023_2623_Fig1_HTML.jpg

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Gynecol Oncol Rep. 2021 Mar 19;36:100750. doi: 10.1016/j.gore.2021.100750. eCollection 2021 May.
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Rhabdomyosarcoma of the cervix in teenagers - Is fertility preservation a feasible option?青少年宫颈横纹肌肉瘤——保留生育功能是否可行?
Gynecol Oncol Rep. 2020 Nov 21;34:100677. doi: 10.1016/j.gore.2020.100677. eCollection 2020 Nov.
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DICER1-associated embryonal rhabdomyosarcoma and adenosarcoma of the gynecologic tract: Pathology, molecular genetics, and indications for molecular testing.
DICER1 相关的胚胎性横纹肌肉瘤和妇科管腔的腺肉瘤:病理学、分子遗传学及分子检测指征。
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Local treatment of rhabdomyosarcoma of the female genital tract: Expert consensus from the Children's Oncology Group, the European Soft-Tissue Sarcoma Group, and the Cooperative Weichteilsarkom Studiengruppe.女性生殖道横纹肌肉瘤的局部治疗:来自儿童肿瘤学组、欧洲软组织肉瘤组和软组织肉瘤合作研究组的专家共识。
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Recurrent Cervical Sarcoma Botryoides in a 3-Year-Old Female: Approach in a Limited Resource Setting.一名3岁女性复发性宫颈葡萄状肉瘤:资源有限情况下的处理方法
Am J Case Rep. 2019 Jun 14;20:838-843. doi: 10.12659/AJCR.915608.
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Second primary rhabdomyosarcoma of the uterine cervix presenting with synchronous ovarian Sertoli-Leydig cell tumor: An illustrative case of DICER1 syndrome.宫颈继发性原发性横纹肌肉瘤伴同步性卵巢支持-间质细胞瘤:DICER1综合征的一个典型病例
Gynecol Oncol Rep. 2018 Jun 15;25:94-97. doi: 10.1016/j.gore.2018.06.008. eCollection 2018 Aug.
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