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源自子宫腺肌病的黏液性癌:一例报告。

Mucinous carcinoma originating from uterine adenomyosis: a case report.

机构信息

Department of Obstetrics and Gynecology, Iida Municipal Hospital, 438 Yawatamachi, Iida, 395-8502, Japan.

Department of Pathology, Iida Municipal Hospital, 438 Yawatamachi, Iida, 395-8502, Japan.

出版信息

J Med Case Rep. 2023 Feb 6;17(1):36. doi: 10.1186/s13256-023-03772-w.

DOI:10.1186/s13256-023-03772-w
PMID:36740698
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC9900899/
Abstract

BACKGROUND

Uterine adenomyosis is rarely a precursor of malignant tumors, but the most frequent histological subtype is endometrioid carcinoma. We observed a rare case of mucinous carcinoma originating from uterine adenomyosis.

CASE PRESENTATION

A 63-year-old Japanese woman presented to our hospital with lower abdominal pain. She had no atypical genital bleeding. Ultrasound demonstrated thickening of the entire uterine wall, but the endometrium was not thick. Magnetic resonance imaging demonstrated an enlarged uterus with thickening of the entire uterine wall, suggesting adenomyosis. On the basis of the specimen of endocervical curettage, adenocarcinoma originating from the endometrium was suspected. Total abdominal hysterectomy and bilateral salpingo-oophorectomy were performed to confirm the diagnosis. Macroscopically, the resected enlarged uterus had no nodules and exudation of mucin was observed from the cut surface of the thickened myometrium. The surface of the endometrium was smooth. On histological examination, mucinous carcinoma invaded almost the entire myometrium. Adenomyotic lesions were distributed focally in the uterine wall, and transition from adenomyotic glandular epithelium to mucinous carcinoma was detected within several foci. Although adenocarcinoma cells proliferated adjacent to the endometrium, the primary endometrial epithelium was atrophic without atypia. Throughout the myometrium, the mucinous carcinoma cells proliferated and floated in dilated lymph vessels with abundant mucin pools. We diagnosed this case as mucinous carcinoma originating from adenomyosis. Although the patient received 11 courses of intravenous adjuvant chemotherapy, she died of disease 18 months after the first operation.

CONCLUSION

As only one case of mucinous carcinoma originating from adenomyosis has been reported to date, this is the second case report of mucinous carcinoma. Moreover, an abnormal manner of proliferation with marked lymphatic permeation of the tumor cells throughout the myometrium was observed.

摘要

背景

子宫腺肌病很少是恶性肿瘤的前兆,但最常见的组织学亚型是子宫内膜样癌。我们观察到一例罕见的源自子宫腺肌病的黏液腺癌病例。

病例介绍

一名 63 岁日本女性因下腹疼痛就诊于我院。她没有出现非典型的生殖器出血。超声检查显示整个子宫壁增厚,但子宫内膜不厚。磁共振成像显示子宫增大,整个子宫壁增厚,提示腺肌病。基于宫颈刮宫标本,怀疑来源于子宫内膜的腺癌。行全子宫切除术和双侧输卵管卵巢切除术以明确诊断。大体上,切除的增大子宫无结节,增厚的子宫肌层切面可见黏液渗出。子宫内膜表面光滑。组织学检查显示,黏液性腺癌几乎侵犯整个子宫肌层。腺肌病病变局限分布于子宫壁内,在数个病灶中检测到从腺肌病腺上皮向黏液腺癌的转变。尽管腺癌细胞在子宫内膜旁增殖,但原发性子宫内膜上皮萎缩,无异型性。在整个子宫肌层中,黏液癌细胞增殖并漂浮在富含黏液池的扩张淋巴管中。我们诊断该病例为源自腺肌病的黏液腺癌。尽管患者接受了 11 个疗程的静脉辅助化疗,但她在首次手术后 18 个月死于疾病。

结论

由于迄今为止仅报道了一例源自腺肌病的黏液腺癌病例,因此这是第二例黏液腺癌病例报告。此外,还观察到肿瘤细胞以异常方式增殖,并伴有明显的淋巴管渗透整个子宫肌层。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/fd63/9900899/c2fed800ecab/13256_2023_3772_Fig3_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/fd63/9900899/c1523bb87398/13256_2023_3772_Fig1_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/fd63/9900899/fd46543d26a8/13256_2023_3772_Fig2_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/fd63/9900899/c2fed800ecab/13256_2023_3772_Fig3_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/fd63/9900899/c1523bb87398/13256_2023_3772_Fig1_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/fd63/9900899/fd46543d26a8/13256_2023_3772_Fig2_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/fd63/9900899/c2fed800ecab/13256_2023_3772_Fig3_HTML.jpg

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本文引用的文献

1
Uterine Adenosarcoma Originating in Adenomyosis: Report of an Extremely Rare Phenomenon and Review of Published Literature.源自子宫腺肌病的子宫腺肉瘤:一种极其罕见现象的报告,并复习文献。
Int J Gynecol Pathol. 2021 Jul 1;40(4):342-348. doi: 10.1097/PGP.0000000000000709.
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The clinicopathological characteristics and survival outcomes of endometrial carcinoma coexisting with or arising in adenomyosis: A pilot study.子宫内膜癌合并或源自于子宫腺肌病的临床病理特征和生存结局:一项初步研究。
Sci Rep. 2020 Apr 6;10(1):5984. doi: 10.1038/s41598-020-63065-w.
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Mucinous endometrial cancer: Clinical study of the eleven cases.
黏液性子宫内膜癌:11例临床研究
North Clin Istanb. 2019 Jul 3;7(1):60-64. doi: 10.14744/nci.2019.17048. eCollection 2020.
4
A Rare Case of Intramural Müllerian Adenosarcoma Arising from Adenomyosis of the Uterus.一例罕见的源于子宫腺肌病的壁间苗勒管腺肉瘤病例。
J Pathol Transl Med. 2017 Jul;51(4):433-440. doi: 10.4132/jptm.2017.06.11. Epub 2017 Jun 29.
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Endometrial cancer arising in adenomyosis versus endometrial cancer coexisting with adenomyosis: are these two different entities?子宫腺肌病中发生的子宫内膜癌与合并子宫腺肌病的子宫内膜癌:这是两种不同的实体吗?
Arch Gynecol Obstet. 2017 Jun;295(6):1459-1468. doi: 10.1007/s00404-017-4375-z. Epub 2017 Apr 25.
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Serous carcinoma arising from uterine adenomyosis/adenomyotic cyst of the cervical stump: a report of 3 cases.宫颈残端子宫腺肌病/腺肌病囊肿来源的浆液性癌:3例报告
Diagn Pathol. 2016 Jun 4;11(1):46. doi: 10.1186/s13000-016-0496-0.
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Carcinosarcoma arising from uterine adenomyosis: A case report.子宫腺肌病并发癌肉瘤:一例报告
J Obstet Gynaecol Res. 2016 Mar;42(3):358-62. doi: 10.1111/jog.12901. Epub 2015 Dec 10.
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Clear cell adenocarcinoma arising from adenomyotic cyst: A case report and literature review.子宫腺肌病囊肿起源的透明细胞腺癌:一例报告及文献复习
J Obstet Gynaecol Res. 2016 Feb;42(2):217-23. doi: 10.1111/jog.12866. Epub 2015 Nov 4.
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Müllerian Adenosarcoma: A Malignant Progression of Adenomyosis? Pictorial Review With Multimodality Imaging.苗勒管腺肉瘤:子宫腺肌病的恶性进展?多模态成像的影像学综述
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The association between endometriosis and ovarian cancer: a review of histological, genetic and molecular alterations.子宫内膜异位症与卵巢癌的关联:组织学、遗传学和分子改变的综述。
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