保守手术治疗的10例Ⅰ期胎盘部位滋养细胞肿瘤病例报告及文献复习

doi:10.19401/j.cnki.1007-3639.2023.09.006

中国癌症杂志 ›› 2023, Vol. 33 ›› Issue (9): 857-865.doi: 10.19401/j.cnki.1007-3639.2023.09.006

保守手术治疗的10例Ⅰ期胎盘部位滋养细胞肿瘤病例报告及文献复习

陈金娟¹(), 王星然¹, 李文质¹, 程煜¹, 孙艺华², 陶祥², 马凤华³, 孙莉⁴, 赵洪波⁵^,⁶, 鹿欣¹()

1.复旦大学附属妇产科医院妇瘤科，上海 200090
2.复旦大学附属妇产科医院病理科，上海 200090
3.复旦大学附属妇产科医院放射科，上海 200090
4.复旦大学附属妇产科医院超声科，上海 200090
5.复旦大学附属妇产科医院组织库，上海 200090
6.上海市女性生殖内分泌相关疾病重点实验室，上海 200090

收稿日期:2023-07-31 修回日期:2023-09-07 出版日期:2023-09-30 发布日期:2023-10-01
通信作者: 鹿欣（ORCID: 0000-0001-8054-6940），博士后，主任医师。
作者简介:陈金娟（ORCID：0000-0001-6097-9968），硕士，主治医师。

Conservative surgery in stage I placental site trophoblastic tumor: a report of 10 cases and literature review

CHEN Jinjuan¹(), WANG Xingran¹, LI Wenzhi¹, CHENG Yu¹, SUN Yihua², TAO Xiang², MA Fenghua³, SUN Li⁴, ZHAO Hongbo⁵^,⁶, LU Xin¹()

1. Department of Gynecology, Obstetrics and Gynecology Hospital of Fudan University, Shanghai 200090, China
2. Department of Pathology, Obstetrics and Gynecology Hospital of Fudan University, Shanghai 200090, China
3. Department of Radiology, Obstetrics and Gynecology Hospital of Fudan University, Shanghai 200090, China
4. Department of Ultrasonography, Obstetrics and Gynecology Hospital of Fudan University, Shanghai 200090, China
5. Department of Obstetrics and Gynecology of Shanghai Medical School of Fudan University, Shanghai 200090, China
6. Shanghai Key Laboratory of Female Reproductive Endocrine Related Diseases, Shanghai 200090, China

Received:2023-07-31 Revised:2023-09-07 Published:2023-09-30 Online:2023-10-01
Contact: LU Xin.

摘要/Abstract

摘要：

背景与目的：胎盘部位滋养细胞肿瘤（placental site trophoblastic tumor，PSTT）是罕见的妇科恶性肿瘤，由于PSTT在子宫内独特的浸润方式和对化疗不敏感特性，全子宫切除是首选的手术方式。本研究旨在探讨Ⅰ期PSTT患者保守手术治疗的可行性和安全性。方法：纳入2015年1月—2021年12月复旦大学附属妇产科医院收治的Ⅰ期PSTT病例，同时分别以“placental site trophoblastic tumor”和“case”、“胎盘部位滋养细胞肿瘤”和“例”为关键词在PubMed和中国知网（CNKI）中检索1990年1月—2021年12月发表的接受保守手术治疗的Ⅰ期PSTT个案报道和系统病例报告，收集患者的临床病理学资料，并进行回顾性分析。结果：共纳入复旦大学附属妇产科医院收治的符合标准的病例10例，中位年龄为27岁，最常见的症状为不规则阴道出血（70.0%），中位诊断距离前次妊娠时间间隔（interval since antecedent pregnancy，ISAP）为14.5个月，中位β-绒毛膜促性腺激素（β-human chorionic gonadotrophin，β-hCG）水平为124.51 mU/mL，病灶直径为0.8 ~ 8.0 cm。复旦大学附属妇产科医院收治的10例患者经初始治疗后均获得完全缓解，平均随访时间为48.1个月，均未复发。3例患者治疗后自然受孕，其中2例足月妊娠分娩，1例因计划外妊娠行人工流产。文献复习PSTT病例显示出类似的临床病理学分布特征和疾病转归。结论：保守手术可以作为部分Ⅰ期PSTT患者的一种替代治疗方法，但需更多研究提供证据。

关键词: 胎盘部位滋养细胞肿瘤, 保守手术, 预后

Abstract:

Background and purpose: Placental site trophoblastic tumor (PSTT) is a rare type of malignant tumor. Because of its unique mode of invasion in the uterus and its insensitivity to chemotherapy, total hysterectomy is the primary recommendation. The purpose of this study was to investigate the feasibility and safety of conservative surgical treatment in patients with stage Ⅰ PSTT. Methods: The patients with stage Ⅰ PSTT admitted to Obstetrics and Gynecology Hospital of Fudan University from January 2015 to December 2021 were included, and those published on Pubmed and China National Knowledge Infrastructure (CNKI) from January 1990 to December 2021 were searched with the keywords of “placental site trophoblastic tumor” and “case”, “placental trophoblastic tumor” and “case” respectively. The clinicopathological data of the patients were collected and retrospectively analyzed. Results: A total of 10 cases admitted to Obstetrics and Gynecology Hospital of Fudan University were enrolled. The median age was 27 years. The most common symptom was irregular vaginal bleeding (70.0%). The median time of interval since antecedent pregnancy (ISAP) was 14.5 months. The median level of β-human chorionic gonadotrophin (β-hCG) was 124.51 mU/mL, and the diameter of the focus was 0.8-8.0 cm. All 10 patients admitted to Obstetrics and Gynecology Hospital of Fudan University achieved complete remission after initial treatment. The average follow-up time was 48.1 months and there was no recurrence. Three patients became pregnant naturally after treatment, including 2 cases of full-term pregnancy and delivery and 1 case of induced abortion because of unplanned pregnancy. Literature review of PSTT cases showed similar clinicopathological distribution and disease outcome. Conclusion: Conservative surgery could be an alternative choice for selected patients with stage Ⅰ PSTT, but more research is needed to provide evidence.

Key words: Placental site trophoblastic tumor, Conservative surgery, Prognosis

中图分类号:

R737.33

陈金娟, 王星然, 李文质, 程煜, 孙艺华, 陶祥, 马凤华, 孙莉, 赵洪波, 鹿欣. 保守手术治疗的10例Ⅰ期胎盘部位滋养细胞肿瘤病例报告及文献复习[J]. 中国癌症杂志, 2023, 33(9): 857-865.

CHEN Jinjuan, WANG Xingran, LI Wenzhi, CHENG Yu, SUN Yihua, TAO Xiang, MA Fenghua, SUN Li, ZHAO Hongbo, LU Xin. Conservative surgery in stage I placental site trophoblastic tumor: a report of 10 cases and literature review[J]. China Oncology, 2023, 33(9): 857-865.

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参考文献 35

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Case	Age	Gravidity	Parity	Antecedent pregnancy	ISAP/month	Chief Complaint	β-hCG/(mU·mL^-1)
1	27	1	1	Term	12	Irregular vaginal bleeding	88.89
2	24	2	1	Term	12	Irregular vaginal bleeding	1 120.00
3	28	2	1	Term	14	Irregular vaginal bleeding	42.31
4	29	3	1	Hydatidiform mole	15	Elevated hCG	160.13
5	28	1	1	Term	28	Irregular vaginal bleeding	0.19
6	27	2	0	Abortion	30	Elevated hCG	363.90
7	28	2	0	Abortion	15	Irregular vaginal bleeding	2 281.00
8	27	1	1	Term	6	Elevated hCG	8.76
9	24	3	0	Abortion	17	Irregular vaginal bleeding	61.66
10	25	3	2	Term	8	Irregular vaginal bleeding	25.60

Case	Lesion size before surgery/cm	Lesion site	Lesion echo	Lesion border	Blood flow
1	8.0×5.5×6.9	Uterine cavity and posterior wall	Locally anechoic	Almost involving the uterine serous layer	++
2	5.6×5.0	Uterine cavity	Hypoechoic	Unclear border with the myometrium	+++
3	2.6×1.8×2.5	Uterine cavity	Iso- and hyper- echoic	Unclear border with the myometrium	+
4	4.1×4.2×3.4	Anterior wall	Hypoechoic	Almost involving the uterine serous layer	+++
5	0.8×0.8×0.7	Anterior wall	Iso- and hypoechoic	Close to the uterine cavity	±
6	3.9×3.3×3.2	Anterior wall	Iso echoic	3 mm to the uterine serous layer	++
7	4.9×4.6×4.8	Anterior wall	Non-uniform	Protruding into the uterine cavity	+++
8	NA	NA	NA	NA	NA
9	1.1×1.0×0.9	Uterine cavity	Iso echoic	Unclear border with the myometrium	+
10	2.4×1.5×2.4	Uterine cavity	Non-uniform	Unclear border with the myometrium	++

Case	Pathologic high-risk factors	Ki-67 proliferation index/%	Vascular remodeling
1	Necrosis and high mitosis	30	Yes
2	No	20	NA
3	No	NA	NA
4	Necrosis and high mitosis	30	Yes
5	No	5	No
6	High mitosis	20	Yes
7	High mitosis	20	Yes
8	No	30	NA
9	High mitosis	5	Yes
10	necrosis	20	NA

Case	Surgery			Chemo-therapy	Follow up/month	Pregnancy after treatment
Case	Diagnostic surgery	Therapeutic surgery	Surgery during follow up	Chemo-therapy	Follow up/month	Pregnancy after treatment
1	LULR^ab	No	No	EMA-EP*3	79-NED	0-fear of recurrence
2	LULR and left ovarian cystectomy^bc	No	No	EMA-EP*2	87-NED	1-full term delivery
3	LULR^b	No	Curettage^b	EMA-EP*2	87-NED	1-full term delivery
4	LULR and left ovarian cystectomy^a	No	Curettage and intrauterine adhesion separation^b	EMA-EP*3	45-NED	0-fear of recurrence
5	LULR^b	LULR^a	Curettage and intrauterine adhesion separation^b	No	44-NED	0-fear of recurrence
6	D&C	LULR^c	Intrauterine adhesion separation and IUD implantation^b	EMA-EP*4	46-NED	0-preparing for pregnancy
7	D&C	LULR^a	No	EP1^d, EMA-EP(6^d+2)	23-NED	0-hypomenorrhea
8	LULR^b	No	Curettage^b	No	32-NED	0-hypomenorrhea
9	D&C	LULR and IUD implantation^a	Removal of IUD, separation of cervical adhesion	No	26-NED	0-fear of recurrence
10	LULR^b	No	Curettage and intrauterine adhesion separation^b	No	12-NED	1-abortion

Characteristic	Case n(%)	Range	x±s/median, Q1-Q3	Characteristic	Case n(%)	Range	x±s/median, Q1-Q3
Chief complaint	34			Necrosis and hemorrhage	19
Irregular vaginal bleeding	18 (52.9)			Yes	7 (36.8)
Menopause	5 (14.7)			No	12 (63.2)
Irregular vaginal bleeding and menopause	3 (8.8)			Mitosis index/10HPF	22
Others	8 (23.5)			≤5	17 (77.3)
Gravidity	26			>5	5 (22.7)
1	12 (46.2)			Ki-67 proliferation index	22
2	5 (19.2)			≤10%	16 (72.7)
3	6 (23.1)			10%-≤30%	4 (18.2)
≥4	3 (11.5)			>30%	2 (9.1)
Parity	26			Surgery	40
0	8 (28.6)			Curettage	16 (40.0)
1	16 (61.5)			Local uterine lesion resection	20 (50.0)
2	2 (7.7)			Unknown	4 (10.0)
Antecedent pregnancy	37			Chemotherapy	40
Term	19 (51.4)			Before surgery	1 (2.5)
Abortion	13 (35.1)			After surgery	15 (37.5)
Hydatidiform mole	5 (13.5)			Both before and after surgery	7 (17.5)
ISAP/month	36	0-29	6, 2-11	Without chemotherapy	17 (42.5)
<12	28 (77.8)			Initial response	40
12-24	6 (16.7)			Remission	36 (90.0)
≥24	2 (5.6)			Persistence	4 (10.0)
Serum β-hCG/(mU·mL^-1)	29	2-5 775	120.3, 53.0-689.5	Follow up/month	40	2-144	36.3, 14.5-49.0
<10²	13 (44.8)			Outcome of follow up	40
10²-10³	10 (34.5)			No evidence of disease	40 (100.0)
10³-10⁴	5 (17.2)			Relapse	0 (0.0)
≥10⁴	1 (3.4)			Pregnant outcome	9
Tumor size/cm	10	0-3.6	2.1±1.3	Term	6
<3	7 (70.0)			Abortion	2
≥3	3 (30.0)			Pregnant	1
Invasion depth	12
Superficial	8 (66.7)
Deep	4 (33.3)

保守手术治疗的10例Ⅰ期胎盘部位滋养细胞肿瘤病例报告及文献复习

Conservative surgery in stage I placental site trophoblastic tumor: a report of 10 cases and literature review

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参考文献 35

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Characteristic	Case A	Case B	Case C	Case D
Age/year	30	24	33	29
Gravidity	1	NA	3	4
Parity	0	NA	1	2
Chief complaint	4	1	4	1
Antecedent pregnancy	3	2	1	2
ISAP/month	2	2	18	3.5
β-HCG/(mU·mL-1)	11 339	NA	NA	80
Tumor size/cm	2.5	NA	NA	2.9
Surgery
Initial surgery	LULR	D&C	D&C	LULR
Hysterectomy	Yes	No	Yes	Yes
Chemotherapy
Before surgery	MTX/CF4, EMA-CO5.5, GC*2	No	No	No
After surgery	(TP-TE)*2.5	No	No	No
Invasion depth	NA	NA	Superficial	Deep
Hemorrhage and necrosis	NA	No	No	Yes
Mitosis index/10HPF	5-7	Sparse	NA	8
Ki-67 proliferation index	NA	NA	NA	>25%
Follow up/month	30	36	6	33
Outcome of follow up	NED	LWD	NED	NED

[1]	金奕滋, 林明曦, 张剑. 乳腺癌原发灶与肝转移灶受体表达差异研究[J]. 中国癌症杂志, 2023, 33(9): 834-843.
[2]	吴晗, 杨章孺, 冯雯, 曾琬琴, 郭金栋, 李洪选, 王常禄, 王家明, 吕长兴, 张琴, 余雯, 蔡旭伟, 傅小龙. 多原发性早期肺癌立体定向放射治疗后疗效及患者预后分析[J]. 中国癌症杂志, 2023, 33(9): 844-856.
[3]	孙洋, 王炼, 赵萌, 张小凤, 耿志军, 王月月, 宋雪, 左芦根, 李静, 胡建国. 胃癌中FKBP1A高表达的预后价值及其靶向PI3K/AKT对糖代谢的调控作用[J]. 中国癌症杂志, 2023, 33(8): 726-739.
[4]	姜琳, 刘绮颖, 贾利晴, 张静, 常恒, 薛田, 任敏, 柏乾明, 朱晓丽, 周晓燕. 胶质瘤中MGMT甲基化状态及其临床意义的回顾性研究[J]. 中国癌症杂志, 2023, 33(8): 740-750.
[5]	王若曦, 吉芃, 龚悦, 陈盛. HER2低表达乳腺癌新辅助化疗效果及其预后特征：一项单中心回顾性研究[J]. 中国癌症杂志, 2023, 33(7): 686-692.
[6]	左学良, 陈志强, 董润雨, 王智雄, 蔡娟. 联合检测LDHA和PD-L1在晚期胃癌PD-1抑制剂疗效预测及预后评估中的价值[J]. 中国癌症杂志, 2023, 33(5): 460-468.
[7]	陈玉光, 孙晓, 毕钊, 邱鹏飞, 段宝伟, 樊庆达, 王永胜. 乳腺癌内乳前哨淋巴结活检评估预后及指导个体化内乳照射：一项长期随访研究[J]. 中国癌症杂志, 2023, 33(2): 142-151.
[8]	陈瑛瑶, 储香玲, 俞昕, 苏春霞. 免疫检查点抑制剂疗效相关预测模型的研究进展[J]. 中国癌症杂志, 2023, 33(1): 61-70.
[9]	高鹤丽, 徐近, 虞先濬. 2021年胰腺神经内分泌肿瘤诊治的新进展[J]. 中国癌症杂志, 2022, 32(9): 772-778.
[10]	张日虹, 郭宇, 梁贇, 陈洛海, 陈洁, 王玮. 辅助化疗对胃神经内分泌癌患者预后的意义[J]. 中国癌症杂志, 2022, 32(9): 807-817.
[11]	曹孟轩, 胡灿, 张延强, 黄灵, 杜义安, 于鹏程, 章若岚, 徐志远, 程向东. 胃癌根治术后早期复发转移的危险因素分析[J]. 中国癌症杂志, 2022, 32(7): 588-595.
[12]	钱瑶(综述), 刘锋(审校). 乳腺癌新辅助内分泌治疗的研究进展及展望[J]. 中国癌症杂志, 2022, 32(7): 643-649.
[13]	刘彦权, 胡晓梅, 殷悦, 林霖, 沈建箴, 陈玉婷, 唐焕文. 器官移植术后淋巴组织增生性疾病的临床回顾性研究[J]. 中国癌症杂志, 2022, 32(7): 650-656.
[14]	曲以平, 侯鹏. 转移性儿童甲状腺癌的遗传学特征和预后[J]. 中国癌症杂志, 2022, 32(5): 373-379.
[15]	马光, 区晓敏, 胡超苏, 宋少莉, 杨忠毅. 治疗前¹⁸F-FLT和¹⁸F-FDG PET/CT摄取异质性在局部晚期鼻咽癌疗效预测中的价值[J]. 中国癌症杂志, 2022, 32(4): 309-315.