子宫附腔畸形一例并文献复习

doi:10.12280/gjfckx.20230008

摘要/Abstract

摘要：

子宫附腔畸形（accessory cavitated uterine malformation，ACUM）是一种罕见的苗勒管畸形，其主要临床表现为从初潮开始进行性加重的痛经，需与囊性子宫腺肌病、子宫肌瘤囊性变及其他相关的阻塞性苗勒管畸形相鉴别。手术是其主要治疗方案。报告1例青少年女性患者，痛经渐进加重，伴恶心呕吐，左髂窝痛，需服止痛药。2022年8月盆腔三维超声示：宫腔形态基本正常，子宫左侧壁不均质结节回声28 mm×26 mm×20 mm，内见内膜回声11 mm×10 mm×7 mm，疑囊性局灶性子宫腺肌病或ACUM，行腹腔镜探查术，见子宫外观正常，其左前壁、圆韧带前下方稍饱满，于饱满处切开肌壁，见一组织较苍白，质韧的包块，完整切除送检，经病理确诊为ACUM。随访术后症状消失。

关键词: 子宫附腔畸形, 子宫, 先天畸形, 苗勒管, 痛经, 病例报告

Abstract:

Accessory cavitated uterine malformation(ACUM) is a rare form of Müllerian anomaly, usually presents with progressive and aggravating dysmenorrhea from menarche. It should be distinguished from cystic adenomyoma, degenerating cystic leiomyoma and the other form of obstructive Müllerian anomaly. Surgery is its main treatment option. In this study, we present an adolescent female with progressive worsening dysmenorrhea, nausea and vomiting, pain in the left iliac fossa and need to take painkillers. In August 2022, three-dimensional pelvic ultrasound in our hospital showed: basic normal uterine cavity shape, left side wall heterogeneity nodular echo 28 mm×26 mm×20 mm with endometrial echo within 11 mm×10 mm×7 mm, suspected cystic focal uterine adenomyosis or ACUM. Laparoscopic exploration was performed. The appearance of the uterus was normal, the left anterior wall and at the anterior and inferior part of round ligament were slightly full. The uterine wall of slightly full was resected, a pale and tough masses were found and resected completely for examination. ACUM was confirmed by pathology. Symptoms disappeared after follow-up surgery.

Key words: Accessory cavitated uterine malformation, Uterus, Congenital abnormalities, Mullerian ducts, Dysmenorrhea, Case reports

苏丹, 李斯静, 黄晓武, 夏恩兰. 子宫附腔畸形一例并文献复习[J]. 国际妇产科学杂志, 2023, 50(3): 266-270.

SU Dan, LI Si-jing, HUANG Xiao-wu, XIA En-lan. Accessory Cavitated Uterine Malformation: A Case Report and Literature Review[J]. Journal of International Obstetrics and Gynecology, 2023, 50(3): 266-270.

图/表 7

图1 2022年6月14日经阴道直肠子宫双侧附件三维超声图注：图A见宫腔形态基本正常，两侧宫角结构可见，内见内膜回声11 mm×10 mm×7 mm，考虑囊性局灶性子宫腺肌病可能，子宫附腔可能；图B见左侧壁不均质结节回声28 mm×26 mm×20 mm。

图2 阴道内镜所见注：阴道内镜下见宫腔形态正常，双侧宫角及双侧输卵管开口可见，箭头所示为双侧输卵管开口。

图3 腹腔镜所见注：腹腔镜探查见子宫大小外观轮廓正常，子宫左前壁稍饱满，双侧附件大小外观正常。图A红色箭头所示为子宫左前壁稍饱满，黄色箭头为左侧圆韧带；图B为腹腔镜下行左侧子宫附腔包块切除，红色箭头所示为包块，黄色箭头所示为子宫肌壁；图C可见腹腔镜下切除组织较苍白质韧，蓝色箭头所示为包块，红色箭头所示为子宫肌壁。

图4 大体标本注：剖视包块切面见内少许似子宫内膜样组织及少许暗褐色液体，白色箭头所示为附腔。

图5 术后病理检查图（HE染色×10）注：见平滑肌组织一侧附少许子宫内膜并见少许含铁血黄素，平滑肌组织中局灶见少许子宫内膜腺体及间质。

图6 2022年9月10日复查经直肠子宫双侧附件三维超声图

图7 囊性子宫腺肌病、Robert子宫、单角子宫伴近端残角子宫伴功能性子宫内膜示意图注：图A为囊性子宫腺肌病示意图，表现为子宫肌壁间囊性包块，边界欠清，周围可见腺肌病病灶；图B为Robert子宫示意图，表现为一半为不交通的宫腔，单侧宫腔积血，一半为单角子宫，子宫底外形正常；图C为单角子宫伴近端残角子宫伴功能性子宫内膜示意图，单角子宫与对侧残角子宫宫底之间有凹陷，残角子宫内含有内膜，但与单角子宫腔不相通。

参考文献 22

[1]	Acién P, Bataller A, Fernández F, et al. New cases of accessory and cavitated uterine masses(ACUM): a significant cause of severe dysmenorrhea and recurrent pelvic pain in young women[J]. Hum Reprod, 2012, 27(3):683-694. doi: 10.1093/humrep/der471. doi: 10.1093/humrep/der471
[2]	Tokgoz VY, Tekin AB. A rare case of the new entity of müllerian anomalies mimicking the noncommunicating rudimentary cavity with hemi-uterus: accessory cavitated uterine mass[J]. Fertil Steril, 2022, 117(3):646-648. doi: 10.1016/j.fertnstert.2021.11.028. doi: 10.1016/j.fertnstert.2021.11.028 pmid: 35058046
[3]	Peters A, Rindos NB, Guido RS, et al. Uterine-sparing Laparoscopic Resection of Accessory Cavitated Uterine Masses[J]. J Minim Invasive Gynecol, 2018, 25(1):24-25. doi: 10.1016/j.jmig.2017.06.001. doi: 10.1016/j.jmig.2017.06.001
[4]	Oliver J. An accessory uterus distended with menstrual fluid enucleated from the substance of the right broad ligament[J]. Lancet, 1912, 179(4633):1609. doi: 10.1016/S0140-6736(00)51005-8. doi: 10.1016/S0140-6736(00)51005-8
[5]	Tamura M, Fukaya T, Takaya R, et al. Juvenile adenomyotic cyst of the corpus uteri with dysmenorrhea[J]. Tohoku J Exp Med, 1996, 178(3):339-344. doi: 10.1620/tjem.178.339. doi: 10.1620/tjem.178.339 pmid: 8727716
[6]	Potter DA, Schenken RS. Noncommunicating accessory uterine cavity[J]. Fertil Steril, 1998, 70(6):1165-1166. doi: 10.1016/s0015-0282(98)00380-x. doi: 10.1016/s0015-0282(98)00380-x pmid: 9848312
[7]	Takeuchi H, Kitade M, Kikuchi I, et al. Diagnosis, laparoscopic management, and histopathologic findings of juvenile cystic adenomyoma: a review of nine cases[J]. Fertil Steril, 2010, 94(3):862-868. doi: 10.1016/j.fertnstert.2009.05.010. doi: 10.1016/j.fertnstert.2009.05.010 pmid: 19539912
[8]	Acién P, Acién M, Fernández F, et al. The cavitated accessory uterine mass: a Müllerian anomaly in women with an otherwise normal uterus[J]. Obstet Gynecol, 2010, 116(5):1101-1109. doi: 10.1097/AOG.0b013e3181f7e735. doi: 10.1097/AOG.0b013e3181f7e735 pmid: 20966695
[9]	Rackow BW. Accessory cavitated uterine mass: a new müllerian anomaly?[J]. Fertil Steril, 2022, 117(3):649-650. doi: 10.1016/j.fertnstert.2022.01.006. doi: 10.1016/j.fertnstert.2022.01.006 pmid: 35105449
[10]	Paul PG, Chopade G, Das T, et al. Accessory Cavitated Uterine Mass: A Rare Cause of Severe Dysmenorrhea in Young Women[J]. J Minim Invasive Gynecol, 2015, 22(7):1300-1303. doi: 10.1016/j.jmig.2015.06.007. doi: 10.1016/j.jmig.2015.06.007
[11]	Pfeifer SM, Attaran M, Goldstein J, et al. ASRM müllerian anomalies classification 2021[J]. Fertil Steril, 2021, 116(5):1238-1252. doi: 10.1016/j.fertnstert.2021.09.025. doi: 10.1016/j.fertnstert.2021.09.025 pmid: 34756327
[12]	Liang YJ, Hao Q, Wu YZ, et al. Uterus-like mass in the left broad ligament misdiagnosed as a malformation of the uterus: a case report of a rare condition and review of the literature[J]. Fertil Steril, 2010, 93(4):1347. e13-e16. doi: 10.1016/j.fertnstert.2009.10.040. doi: 10.1016/j.fertnstert.2009.10.040
[13]	Azuma Y, Taniguchi F, Wibisono H, et al. A Case Report of an Accessory and Cavitated Uterine Mass Treated with Total Laparoscopic Hysterectomy[J]. Yonago Acta Med, 2021, 64(2):207-209. doi: 10.33160/yam.2021.05.004. doi: 10.33160/yam.2021.05.004 pmid: 34025196
[14]	Acién P, Acién M. Accessory and cavitated uterine mass versus juvenile cystic adenomyoma[J]. F S Rep, 2021, 2(3):357-358. doi: 10.1016/j.xfre.2021.06.006. doi: 10.1016/j.xfre.2021.06.006
[15]	Peyron N, Jacquemier E, Charlot M, et al. Accessory cavitated uterine mass: MRI features and surgical correlations of a rare but under-recognised entity[J]. Eur Radiol, 2019, 29(3):1144-1152. doi: 10.1007/s00330-018-5686-6. doi: 10.1007/s00330-018-5686-6 pmid: 30159623
[16]	Putta T, John R, Simon B, et al. Imaging Manifestations of Accessory Cavitated Uterine Mass-A Rare Mullerian Anomaly[J]. Indian J Radiol Imaging, 2021, 31(3):545-550. doi: 10.1055/s-0041-1735504. doi: 10.1055/s-0041-1735504
[17]	Mollion M, Host A, Faller E, et al. Report of two cases of Accessory Cavitated Uterine Mass (ACUM): Diagnostic challenge for MRI[J]. Radiol Case Rep, 2021, 16(11):3465-3469. doi: 10.1016/j.radcr.2021.07.071. doi: 10.1016/j.radcr.2021.07.071
[18]	Bedaiwy MA, Henry DN, Elguero S, et al. Accessory and cavitated uterine mass with functional endometrium in an adolescent: diagnosis and laparoscopic excision technique[J]. J Pediatr Adolesc Gynecol, 2013, 26(4):e89-e91. doi: 10.1016/j.jpag.2012.11.003. doi: 10.1016/j.jpag.2012.11.003
[19]	Park JC, Kim DJ. Successful laparoscopic surgery of accessory cavitated uterine mass in young women with severe dysmenorrhea[J]. Yeungnam Univ J Med, 2021, 38(3):235-239. doi: 10.12701/yujm.2020.00696. doi: 10.12701/yujm.2020.00696
[20]	Merviel P, Lelievre C, Cambier T, et al. The first ethanol sclerotherapy of an accessory cavitated uterine mass[J]. Clin Case Rep, 2021, 9(1):19-22. doi: 10.1002/ccr3.3371. doi: 10.1002/ccr3.3371
[21]	Naftalin J, Bean E, Saridogan E, et al. Imaging in gynecological disease (21): clinical and ultrasound characteristics of accessory cavitated uterine malformations[J]. Ultrasound Obstet Gynecol, 2021, 57(5):821-828. doi: 10.1002/uog.22173. doi: 10.1002/uog.22173
[22]	Supermaniam S, Thye WL. Diagnosis and laparoscopic excision of accessory cavitated uterine mass in young women: Two case reports[J]. Case Rep Womens Health, 2020, 26:e00187. doi: 10.1016/j.crwh.2020.e00187. doi: 10.1016/j.crwh.2020.e00187