Mesonephric adenocarcinoma of uterine cervix: a case report and literature review

doi:10.3877/cma.j.issn.1673-5250.2024.01.013

Objective

To investigate clinical manifestations, diagnosis, treatment and prognosis of mesonephric adenocarcinoma (MA) of uterine cervix.

Methods

A patient (patient 1) with MA in the Xijing Hospital of Air Force Medical University in December 2021 was selected in this study. Retrospective analysis was used to collect the examination results, main treatment course, prognosis and pathological examination results of patient 1 after admission. With " cervix" " adenocarcinoma of middle kidney" " uterine cervix" and " mesonephric adeno carcinoma" as Chinese and English key words, the related literatures about cervical MA were searched from CNKI, Wanfang Database, VIP Chinese Sci-tech Journal Database and PubMed. The literature retrieval time was set from the establishment of the above databases to February 2023. This study was approved by the Medical Ethics Committee of the Xijing Hospital of Air Force Medical University (Approve No.KY20222112-C-1 C-1), and the informed consent was signed by the patient.

Results

①The first symptom of patient 1 was irregular vaginal bleeding. Imaging examination revealed suspicious uterine cavity and cervical mass, and uterine cavity and cervical biopsy revealed adenocarcinoma. Then, abdominal extensive total hysterectomy, double appendages, greater omentum resection+ pelvic cavity, abdominal paraaortic lymph node dissection+ abdominal lipoma resection were performed, and paclitaxel + carboplatin regimen chemotherapy was performed for 4 courses after operation. There were no signs of recurrence and metastasis after one year of follow-up. ② Review of the literature: According to the literature retrieval strategy set by this study, 25 articles were retrieved involving cervical MA patients, with a total of 27 MA patients. Combined with the patient 1 in this study, there were a total of 28 MA patients. The age of onset ranged from 27 to 77 years, with a median age of 57 years. The most common clinical symptoms in the 28 patients were irregular vaginal bleeding (19/28, 67.9%), including postmenopausal vaginal bleeding.

Conclusions

Cervical MA has a poor prognosis, pathological diagnosis depends on immunohistochemical staining, and treatment and prognosis still need a large number of cases to accumulate. Gene detection can provide a basis for targeted treatment of renal adenocarcinoma in cervix.

Key words: Mesonephros, Adenocarcinoma, Pathological conditions, signs and symptoms, Clinical protocols, Immunohistochemistry, Prognosis, Women

图1 宫颈中肾腺癌患者1(女性，67岁，G₃P₃)宫颈组织病理学检查结果[图1A：宫颈病变组织HE染色见异形腺体呈不规则腺管状、筛状、乳头状排列，细胞核变圆淡染，可见核仁，间质减少，浸润性生长(HE染色，中倍)；图1B：子宫内膜病变组织HE染色见异形腺体呈不规则腺管状、筛状、乳头状排列，细胞核变圆淡染，可见核仁，间质减少，浸润性生长(HE染色，中倍)；图1C：宫颈病变组织TTF1局灶阳性(TTF1免疫组化染色，中倍)；图1D：宫颈病变组织CD10局灶阳性(CD10免疫组化染色，中倍)；图1E：宫颈病变组织PR呈阴性(PR免疫组化染色，中倍)；图1F：宫颈病变ER呈阴性(ER免疫组化染色，中倍)]注：HE为苏木素-伊红，TTF1为甲状腺转录因子1, PR为孕激素受体，ER为雌激素受体

表1 28例宫颈MA患者的临床特征

患者编号	文献(第一作者，发表年)	例数	年龄(岁)	孕次/产次	临床表现	查体结果	活组织病理学检查结果	临床分期
1	本研究	1	67	G₃P₃	绝经后阴道流血	宫颈萎缩，光滑，宫体大小如孕50 d	宫腔、宫颈腺癌	ⅠB2(FIGO 2018)
2	Yap^[4]，2006	1	54	—	绝经后阴道流血	宫颈息肉	子宫内膜样腺癌	ⅠB1
3	Fukunagam^[5]，2008	1	46	G₁P₁	异常子宫流血	宫颈可见一个外生型脆性肿物	子宫内膜样腺癌	—
4/5	Nomoto^[6]，2012	2	64/64	G₃P₃/G₂P₂	均为绝经后阴道流血	宫颈萎缩/—	均为子宫内膜样癌	ⅠB2/ⅠB1　(FIGO 2009)
6	Anagnostopoulos^[7]，2012	1	64	P₄^a	无症状	—	宫颈无恶性特征，宫颈中肾腺癌	ⅠB1(FIGO 2009)
7	Menons^[8]，2013	1	65	—	绝经后阴道流血	宫颈质硬，宫颈肿物3 cm×3 cm	子宫内膜样腺癌	ⅠB1(FIGO 2009)
8	Abdul-Ghafar^[9]，2013	1	48	G₄P₂	无症状	子宫轻度增大，伴多发肿块，全正常	—	—
9	Meguro^[10]，2013	1	63	—	绝经后阴道出血	—	未分化癌	ⅡA(FIGO 2009)
10	Yeo^[11]，2014	1	52	G₂P₂	—	—	中肾腺癌(肺楔形活检)	ⅣB
11	Tseng^[12]，2014	1	59	G₆P₃	下腹痛伴排尿困难	—	恶性肿瘤	pT3bN1(病理分级)
12	Puljiz^[13]，2016	1	57	—	绝经后阴道流血	宫颈活动性流血	宫颈腺癌	ⅠB2(FIGO 2009)
13	Dierickx^[14]，2016	1	66	—	绝经后阴道流血	—	宫颈锥切提示MA	ⅡB(FIGO 2009)
14	Cavalcanti^[15]，2017	1	59	—	绝经后阴道流血	—	神经内分泌特征低分化癌	ⅠB1(FIGO 2009)
15	Skala^[16]，2019	1	40	—	盆腔脏器脱垂	—	子宫内膜中肾增生	—
16	Montalvo^[17]，2019	1	48	—	无症状	宫颈息肉	中肾腺癌	—
17	Jiang^[18]，2020	1	48	G₂P₁	无症状	宫颈正常	—	Ⅳ
18	Reis-De-Carvalho^[19]，2021	1	60	—	绝经后阴道流血	宫颈萎缩，余正常	组织学类型不明恶性肿瘤浸润，最终提示HSIL	ⅠB1(FIGO 2018)
19	Nili^[20]，2021	1	46	G₄P₂	异常子宫流血	宫颈肥大，光滑	中肾增生	—
20	Devarashetty^[21]，2022	1	73	—	绝经后阴道流血	—	—	ⅠB
21	侯素平^[22]，2013	1	27	—	阴道淋漓流血	宫颈肥大，宫颈口可见乳头状突起	宫颈MA	ⅡB(FIGO 2009)
22	李悦^[23]，2015	1	62	G₂P₂	绝经后阴道流血	宫颈口可见一鱼肉样组织。左侧阴道穹窿变浅	子宫内膜样腺癌	ⅡA1(FIGO 2009)
23	薛丹^[24]，2017	1	—	—	停经31^＋4周，咳嗽加重3 d	宫颈肥大，右下腹内可触及包块，质硬	宫颈MA	Ⅳ
24	夏琴^[25]，2018	1	42	—	不规则阴道流血	—	MA	—
25/26	关会娟^[26] 2019	2	77/51	—/—	绝经后阴道出血/阴道流血	宫颈口见一5 cm光滑肿物脱/宫颈可见一约2 cm赘生物	子宫内膜间质肉瘤/低分化癌，癌肉瘤，滑膜肉瘤	—/—
27	陈玉莹^[27]，2020	1	35	G₃P₁	不规则阴道流血	宫颈增粗，直径约6 cm，呈菜花样	MA	ⅠB2(FIGO 2009)
28	闵锐^[28]，2022	1	49	—	痛经进行性加重4^＋年、尿频1^＋年	子宫颈轻度糜烂，稍肥大	—	—

表1 28例宫颈MA患者的临床特征

患者编号	文献(第一作者，发表年)	例数	年龄(岁)	孕次/产次	临床表现	查体结果	活组织病理学检查结果	临床分期
1	本研究	1	67	G₃P₃	绝经后阴道流血	宫颈萎缩，光滑，宫体大小如孕50 d	宫腔、宫颈腺癌	ⅠB2(FIGO 2018)
2	Yap^[4]，2006	1	54	—	绝经后阴道流血	宫颈息肉	子宫内膜样腺癌	ⅠB1
3	Fukunagam^[5]，2008	1	46	G₁P₁	异常子宫流血	宫颈可见一个外生型脆性肿物	子宫内膜样腺癌	—
4/5	Nomoto^[6]，2012	2	64/64	G₃P₃/G₂P₂	均为绝经后阴道流血	宫颈萎缩/—	均为子宫内膜样癌	ⅠB2/ⅠB1　(FIGO 2009)
6	Anagnostopoulos^[7]，2012	1	64	P₄^a	无症状	—	宫颈无恶性特征，宫颈中肾腺癌	ⅠB1(FIGO 2009)
7	Menons^[8]，2013	1	65	—	绝经后阴道流血	宫颈质硬，宫颈肿物3 cm×3 cm	子宫内膜样腺癌	ⅠB1(FIGO 2009)
8	Abdul-Ghafar^[9]，2013	1	48	G₄P₂	无症状	子宫轻度增大，伴多发肿块，全正常	—	—
9	Meguro^[10]，2013	1	63	—	绝经后阴道出血	—	未分化癌	ⅡA(FIGO 2009)
10	Yeo^[11]，2014	1	52	G₂P₂	—	—	中肾腺癌(肺楔形活检)	ⅣB
11	Tseng^[12]，2014	1	59	G₆P₃	下腹痛伴排尿困难	—	恶性肿瘤	pT3bN1(病理分级)
12	Puljiz^[13]，2016	1	57	—	绝经后阴道流血	宫颈活动性流血	宫颈腺癌	ⅠB2(FIGO 2009)
13	Dierickx^[14]，2016	1	66	—	绝经后阴道流血	—	宫颈锥切提示MA	ⅡB(FIGO 2009)
14	Cavalcanti^[15]，2017	1	59	—	绝经后阴道流血	—	神经内分泌特征低分化癌	ⅠB1(FIGO 2009)
15	Skala^[16]，2019	1	40	—	盆腔脏器脱垂	—	子宫内膜中肾增生	—
16	Montalvo^[17]，2019	1	48	—	无症状	宫颈息肉	中肾腺癌	—
17	Jiang^[18]，2020	1	48	G₂P₁	无症状	宫颈正常	—	Ⅳ
18	Reis-De-Carvalho^[19]，2021	1	60	—	绝经后阴道流血	宫颈萎缩，余正常	组织学类型不明恶性肿瘤浸润，最终提示HSIL	ⅠB1(FIGO 2018)
19	Nili^[20]，2021	1	46	G₄P₂	异常子宫流血	宫颈肥大，光滑	中肾增生	—
20	Devarashetty^[21]，2022	1	73	—	绝经后阴道流血	—	—	ⅠB
21	侯素平^[22]，2013	1	27	—	阴道淋漓流血	宫颈肥大，宫颈口可见乳头状突起	宫颈MA	ⅡB(FIGO 2009)
22	李悦^[23]，2015	1	62	G₂P₂	绝经后阴道流血	宫颈口可见一鱼肉样组织。左侧阴道穹窿变浅	子宫内膜样腺癌	ⅡA1(FIGO 2009)
23	薛丹^[24]，2017	1	—	—	停经31^＋4周，咳嗽加重3 d	宫颈肥大，右下腹内可触及包块，质硬	宫颈MA	Ⅳ
24	夏琴^[25]，2018	1	42	—	不规则阴道流血	—	MA	—
25/26	关会娟^[26] 2019	2	77/51	—/—	绝经后阴道出血/阴道流血	宫颈口见一5 cm光滑肿物脱/宫颈可见一约2 cm赘生物	子宫内膜间质肉瘤/低分化癌，癌肉瘤，滑膜肉瘤	—/—
27	陈玉莹^[27]，2020	1	35	G₃P₁	不规则阴道流血	宫颈增粗，直径约6 cm，呈菜花样	MA	ⅠB2(FIGO 2009)
28	闵锐^[28]，2022	1	49	—	痛经进行性加重4^＋年、尿频1^＋年	子宫颈轻度糜烂，稍肥大	—	—

表2 28例宫颈MA患者的病理学检查结果

患者编号	文献(第一作者，发表年)	例数	肿瘤直径(cm)	免疫组化
患者编号	文献(第一作者，发表年)	例数	肿瘤直径(cm)	EMA	CEA	calretinin	vimentin	ER	PR	GATA3	CD10	PAX8	TTF1	Ki-67
1	本研究		4.0	NA	NA	包膜＋	部分上皮＋	－	－	灶＋	灶＋	＋	灶＋	40%
2	Yap^[4]，2006	1	2.5	＋	－	－	－	－	NA	NA	－	NA	NA	NA
3	Fukunagam^[5]，2008	1	4.0	＋	－	灶＋	－	－	－	NA	－	NA	NA	15%～20%
4/5	Nomoto^[6]，2012	2	5.2/1.5	弥漫＋/　弥漫＋	－/－	－/－	灶＋/弥漫＋	－/－	－/－	均为　NA	弥漫　＋/－	均为　NA	均为　NA	均为　NA
6	Anagnostopoulos^[7]，2012	1	1.5	＋	－	＋	＋	NA	NA	NA	灶＋	NA	－	NA
7	Menons^[8]，2013	1	NA	NA	－	NA	－	－	－	NA	NA	NA	NA	NA
8	Abdul-Ghafar^[9]，2013	1	NA	＋	－	NA	NA	－	－	NA	＋	NA	NA	略高
9	Meguro^[10]，2013	1	1.8	NA	NA	＋	－	－	－	NA	＋	NA	NA	NA
10	Yeo^[11]，2014	1	NA	弥漫＋	NA	－	弥漫＋	－	－	NA	灶＋	NA	－	NA
11	Tseng^[12]，2014	1	6.3	NA	NA	灶＋	弥漫＋	灶＋	－	NA	灶＋	NA	NA	NA
12	Puljiz^[13]，2016	1	5.5	NA	－	NA	弥漫＋	－	－	NA	NA	NA	NA	60%
13	Dierickx^[14]，2016	1	3.0	＋	－	－	＋	－	－	NA	NA	NA	NA	NA
14	Cavalcanti^[15]，2017	1	3.6	NA	NA	<5%	NA	－	－	斑片＋	＋	斑片＋	－	NA
15	Skala^[16]，2019	1	2.7	NA	NA	－	NA	－	－	弥漫＋	＋	NA	NA	10%
16	Montalvo^[17]，2019	1	NA	NA	－	－	NA	－	－	NA	＋	NA	NA	46%
17	Jiang^[18]，2020	1	4	NA	NA	灶＋	NA	NA	NA	NA	＋	NA	NA	NA
18	Reis-De-Carvalho^[19]，2021	1	1.5	NA	＋	＋	－	－	－	NA	NA	NA	NA	NA
19	Nili^[20]，2021	1	4.5	NA	－	灶＋	灶＋	－	－	NA	灶＋	＋	NA	NA
20	Devarashetty^[21]，2022	1	NA	NA	＋	NA	NA	－	－	NA	＋	NA	NA	NA
21	侯素平^[22]，2013	1	3.5	NA	NA	NA	＋	－	－	NA	NA	NA	NA	NA
22	李悦^[23]，2015	1	NA	NA	－	－	＋	－	－	NA	－	NA	NA	＋
23	薛丹^[24]，2017	1	NA	NA	NA	－	NA	－	－	NA	＋	NA	NA	NA
24	夏琴^[25]，2018	1	1.5	NA	－	－	NA	－	－	＋	＋	强＋	NA	NA
25/26	关会娟^[26]，2019	2	5.0/3.0	灶＋/灶＋	－/－	均为　NA	均为弥　漫强＋	－/－	－/－	＋/＋	均为弥　漫强＋	均为弥　漫强＋	均为　NA	20%～60%^a
27	陈玉莹^[27]，2020	1	3	NA	－	－	＋	NA	NA	NA	灶＋	＋	NA	15%
28	闵锐^[28]，2022	1	NA	NA	NA	灶＋	NA	－	－	＋	NA	＋	NA	<5%

表2 28例宫颈MA患者的病理学检查结果

患者编号	文献(第一作者，发表年)	例数	肿瘤直径(cm)	免疫组化
患者编号	文献(第一作者，发表年)	例数	肿瘤直径(cm)	EMA	CEA	calretinin	vimentin	ER	PR	GATA3	CD10	PAX8	TTF1	Ki-67
1	本研究		4.0	NA	NA	包膜＋	部分上皮＋	－	－	灶＋	灶＋	＋	灶＋	40%
2	Yap^[4]，2006	1	2.5	＋	－	－	－	－	NA	NA	－	NA	NA	NA
3	Fukunagam^[5]，2008	1	4.0	＋	－	灶＋	－	－	－	NA	－	NA	NA	15%～20%
4/5	Nomoto^[6]，2012	2	5.2/1.5	弥漫＋/　弥漫＋	－/－	－/－	灶＋/弥漫＋	－/－	－/－	均为　NA	弥漫　＋/－	均为　NA	均为　NA	均为　NA
6	Anagnostopoulos^[7]，2012	1	1.5	＋	－	＋	＋	NA	NA	NA	灶＋	NA	－	NA
7	Menons^[8]，2013	1	NA	NA	－	NA	－	－	－	NA	NA	NA	NA	NA
8	Abdul-Ghafar^[9]，2013	1	NA	＋	－	NA	NA	－	－	NA	＋	NA	NA	略高
9	Meguro^[10]，2013	1	1.8	NA	NA	＋	－	－	－	NA	＋	NA	NA	NA
10	Yeo^[11]，2014	1	NA	弥漫＋	NA	－	弥漫＋	－	－	NA	灶＋	NA	－	NA
11	Tseng^[12]，2014	1	6.3	NA	NA	灶＋	弥漫＋	灶＋	－	NA	灶＋	NA	NA	NA
12	Puljiz^[13]，2016	1	5.5	NA	－	NA	弥漫＋	－	－	NA	NA	NA	NA	60%
13	Dierickx^[14]，2016	1	3.0	＋	－	－	＋	－	－	NA	NA	NA	NA	NA
14	Cavalcanti^[15]，2017	1	3.6	NA	NA	<5%	NA	－	－	斑片＋	＋	斑片＋	－	NA
15	Skala^[16]，2019	1	2.7	NA	NA	－	NA	－	－	弥漫＋	＋	NA	NA	10%
16	Montalvo^[17]，2019	1	NA	NA	－	－	NA	－	－	NA	＋	NA	NA	46%
17	Jiang^[18]，2020	1	4	NA	NA	灶＋	NA	NA	NA	NA	＋	NA	NA	NA
18	Reis-De-Carvalho^[19]，2021	1	1.5	NA	＋	＋	－	－	－	NA	NA	NA	NA	NA
19	Nili^[20]，2021	1	4.5	NA	－	灶＋	灶＋	－	－	NA	灶＋	＋	NA	NA
20	Devarashetty^[21]，2022	1	NA	NA	＋	NA	NA	－	－	NA	＋	NA	NA	NA
21	侯素平^[22]，2013	1	3.5	NA	NA	NA	＋	－	－	NA	NA	NA	NA	NA
22	李悦^[23]，2015	1	NA	NA	－	－	＋	－	－	NA	－	NA	NA	＋
23	薛丹^[24]，2017	1	NA	NA	NA	－	NA	－	－	NA	＋	NA	NA	NA
24	夏琴^[25]，2018	1	1.5	NA	－	－	NA	－	－	＋	＋	强＋	NA	NA
25/26	关会娟^[26]，2019	2	5.0/3.0	灶＋/灶＋	－/－	均为　NA	均为弥　漫强＋	－/－	－/－	＋/＋	均为弥　漫强＋	均为弥　漫强＋	均为　NA	20%～60%^a
27	陈玉莹^[27]，2020	1	3	NA	－	－	＋	NA	NA	NA	灶＋	＋	NA	15%
28	闵锐^[28]，2022	1	NA	NA	NA	灶＋	NA	－	－	＋	NA	＋	NA	<5%

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