12例子宫外低级别子宫内膜间质肉瘤患者的回顾性分析及文献复习

doi:10.19401/j.cnki.1007-3639.2024.04.009

摘要/Abstract

关键词: 子宫外低级别子宫内膜间质肉瘤, 手术方式, 保育治疗, 激素治疗

Key words: Low-grade extrauterine endometrial stromal sarcoma (LGEESS), Surgical approach, Fertility-sparing surgery, Hormonal therapy

中图分类号:

R737.33

史佳璐, 朱婷婷, 陶祥, 马凤华, 林芝, 邵珺, 赵宇清. 12例子宫外低级别子宫内膜间质肉瘤患者的回顾性分析及文献复习[J]. 中国癌症杂志, 2024, 34(4): 418-424.

SHI Jialu, ZHU Tingting, TAO Xiang, MA Fenghua, LIN Zhi, SHAO Jun, ZHAO Yuqing. Retrospective analysis of 12 cases of low grade extrauterine endometrial stromal sarcoma and literature review[J]. China Oncology, 2024, 34(4): 418-424.

图/表 3

表1

12例LGEESS患者的临床特征"

No	Age	Menstruation status	Gravidity	Parity		Dysmenorrhea		Endometriosis	Uterine myoma-related surgery		Initial clinical presentation
1	51	Postmenopausal	3	3		-		+	-		Abdominal pain
2	44	Premenopausal	5	2		-		-	-		Abdominal swelling and pain
3	27	Premenopausal	0	0		+		+	-		-
4	33	Premenopausal	2	1		+		-	-		Urinary retention
5	40	Premenopausal	4	1		+		-	Myomectomy		-
6	50	Postmenopausal	1	1		-		+	Myomectomy + STH		-
7	34	Premenopausal	3	1		-		-	Myomectomy		-
8	50	Premenopausal	3	1		-		+	Myomectomy		Abdominal pain
9	59	Postmenopausal	2	1		+		+	-		Abdominal swelling and pain
10	73	Postmenopausal	2	1		-		+	-		Frequent micturition, abdominal swelling
11	47	Postmenopausal	2	1		-		+	-		Abdominal pain
12	50	Postmenopausal	2	2		+		+	-		Oppression in chest and anhelation
No	Location				Size D/cm		Treatment		PFS t/month	Location of recurrence		Follow-up t/month
1	Bilateral ovary				6		TH + BSO + CT		Unk	Unk		Unk
2	Left ovary, omentum, colon				5.5		TH + BSO + tumor resection + CT		-	-		NED (148)
3	Uterosacral ligament, posterior lobe of broad ligament				4		FSS + HT1		12	Pelvis		NED (97)
4	Retroperitoneum				12		Tumor resection + TH + BSO + ureteroscope + cystoscope + ureteral stenting		-	-		NED (95)
5	Bilateral ovaries, peritoneum, mesentery, intestine				4		Tumor resection + TH + BSO + L + RT + HT2		-	-		NED (74)
6	Left ovary				6		Cervicectomy + BSO + L + omentectomy + CT		-	-		NED (67)
7	Retroperitoneum				6		FSS + HT3		33	Peritoneum		NED (49)
8	Omentum, pelvic				7		Tumor resection + TH + BSO + omentectomy + HT3		8	Abdominopelvic, abdominal wall, ascending colon, duodenum, liver		AWD (40)
9	Bilateral ovaries				11		TH + BSO + HT3		-	-		NED (34)
10	Right ovary, spread to left ovary and uterus				15		TH + BSO + L + HT3		-	-		NED (8)
11	Left ovary, spread to the serous surface of both fallopian tubes and uterus				7		TH + BS + tumor resection + ureteral stenting		15	Bilateral ovaries, peritoneum, omentum, roof of vagina, rectum		NED (25)
12	Abdominopelvic, chest or liver				-		FNA		0	-		DOD (6)

表1

表2

表3

参考文献 20

[1]	ALI R H, ROUZBAHMAN M. Endometrial stromal tumors revisited: an update based on the 2014 WHO classification[J]. J Clin Pathol, 2015, 68(5): 325-332.
[2]	MASAND R P, EUSCHER E D, DEAVERS M T, et al. Endometrioid stromal sarcoma: a clinicopathologic study of 63 cases[J]. Am J Surg Pathol, 2013, 37(11): 1635-1647. doi: 10.1097/PAS.0000000000000083 pmid: 24121169
[3]	OLIVA E, EGGER J F, YOUNG R H. Primary endometrioid stromal sarcoma of the ovary: a clinicopathologic study of 27 cases with morphologic and behavioral features similar to those of uterine low-grade endometrial stromal sarcoma[J]. Am J Surg Pathol, 2014, 38(3): 305-315. doi: 10.1097/PAS.0000000000000145 pmid: 24525500
[4]	WU Y, LI N, ZHANG R, et al. Primary low-grade extrauterine endometrial stromal sarcoma: analysis of 10 cases with a review of the literature[J]. World J Surg Oncol, 2022, 20(1): 17. doi: 10.1186/s12957-021-02474-1 pmid: 35027071
[5]	XIE W M, BI X N, CAO D Y, et al. Primary endometrioid stromal sarcomas of the ovary: a clinicopathological study of 14 cases with a review of the literature[J]. Oncotarget, 2017, 8(38): 63345-63352. doi: 10.18632/oncotarget.18805 pmid: 28968994
[6]	MASAND R P. Unusual presentations of gynecologic tumors: primary, extrauterine, low-grade endometrioid stromal sarcoma[J]. Arch Pathol Lab Med, 2018, 142(4): 536-541. doi: 10.5858/arpa.2017-0241-RA pmid: 29293021
[7]	王富强, 谭改民. 子宫外子宫内膜间质肉瘤6例临床病理分析[J]. 临床与实验病理学杂志, 2017, 33(5): 565-567.
	WANG F Q, TAN G M. Clinicopathological analysis of 6 cases of extrauterine endometrial stromal sarcoma[J]. Chin J Clin Exp Pathol, 2017, 33(5): 565-567.
[8]	SUN L L, ZHAO W, ZHAO Z H, et al. JAZF1, YWHAE and BCOR gene translocation in primary extrauterine low-grade and high-grade endometrial stromal sarcomas[J]. Histopathology, 2022, 80(5): 809-819.
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[11]	WANG Q, ZHAO X, HAN P. Endometrial stromal sarcoma arising in colorectal endometriosis: a case report and review of the literature[J]. Case Rep Obstet Gynecol, 2015, 2015: 534273.
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[13]	LAUFER J, SCASSO S, KIM B, et al. Fertility-sparing management of low-grade endometrial stromal sarcoma[J]. Int J Gynecol Cancer, 2023, 33(7): 1145-1149. doi: 10.1136/ijgc-2023-004448 pmid: 37400123
[14]	PIĄTEK S, SZYMUSIK I, DAŃSKA-BIDZIŃSKA A, et al. Fertility-sparing management may be considered in young women with uterine sarcoma[J]. J Clin Med, 2022, 11(16): 4761.
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[16]	GU Y Z, DUAN N Y, CHENG H X, et al. Fertility-sparing surgeries without adjuvant therapy through term pregnancies in a patient with low-grade endometrial stromal sarcoma: a case report[J]. World J Clin Cases, 2021, 9(4): 983-991.
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[18]	罗祎, 尹楠林, 曹毅, 等. 阴道子宫内膜间质肉瘤5年后局部复发病例临床病理观察(病例报道+文献复习)[J]. 重庆医科大学学报, 2018, 43(3): 473-476.
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[20]	SQALLI HOUSSAINI M, HALOUA M, MOURABITI A Y, et al. A case of ovarian endometrial stromal sarcoma: radiological and histopathological findings[J]. Radiol Case Rep, 2023, 18(10): 3529-3534.

Parameter	Number of patients	Percentage/%	Parameter	Number of patients	Percentage/%
Tumor primary location			Presence of sex cord differentiation	2	16.7
Ovary	7	58.3	Presence of glandular differentiation	1	8.3
Others	5	41.7	Associated endometriosis
Tumor amount			+	7	58.3
1	3	25.0	-	5	41.7
≥2	9	75.0	Immunohistochemical results
Maximum tumor diameter D/cm			ER+	10	83.3
≤5	2	16.7	PR+	10	83.3
>5	10	83.3	CD10+	12	100.0
LVSI			FISH
+	0	0	JAZF1⁺	3	25.0
-	12	100.0	JAZF1^-	2	16.7
Morphologic patterns			YWHAE^-	1	8.3
Presence of smooth muscle differentiation	2	16.7	BCOR^-	2	16.7

Characteristic	Case n	Percentage/%	Characteristic	Case n	Percentage/%
Age/year			Associated endometriosis
≤50	67	60.4	+	35	-
>50	44	39.6	-	5	-
Initial clinical presentation	85		Immunohistochemical results
Abdominal pain or swelling	37	43.5	ER⁺	61	87.1
Vaginal bleeding	10	11.8	PR⁺	62	88.6
None	26	30.6	CD10⁺	66	93.0
CA12-5			FISH
Elevated	16	-	JAZF1⁺	4	-
Normal	5	-	JAZF1^-	1	-
Tumor primary location			YWHAE^-	1	-
Ovary	66	59.5	BCOR^-	0	-
Others	45	40.5	Initial surgery	110
Tumor amount			TH+BSO	57	51.8
1	55	58.5	Tumor section	18	16.4
≥2	39	41.5	Lateral adnexectomy	10	9.1
Maximum tumor diameter D/cm			Lymphadenectomy	6	5.5
≤5	25	27.5	HT	28	-
>5	66	72.5

12例子宫外低级别子宫内膜间质肉瘤患者的回顾性分析及文献复习

Retrospective analysis of 12 cases of low grade extrauterine endometrial stromal sarcoma and literature review

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