子宫切除术后15年并发阴道残端子宫内膜异位症一例
Endometriosis of Vaginal Stump 15 Years after Hysterectomy: A Case Report
通讯作者: 杨永秀,E-mail:yongxiuyang@163.com
本文编辑: 王琳
收稿日期: 2022-05-5
| 基金资助: |
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Corresponding authors: YANG Yong-xiu, E-mail:yongxiuyang@163.com
Received: 2022-05-5
子宫内膜异位症(endometriosis,EMs)是女性常见的慢性炎症性疾病,指子宫内膜的腺体和间质出现在子宫腔以外的部位,常见于卵巢、盆腔腹膜表面或其他盆腔组织。报告1例子宫肌瘤行子宫切除术后15年发生阴道残端子宫内膜异位症的病例,患者48岁,因不规则阴道出血伴腹痛3年、加重2个月就诊,全腹增强CT发现左侧髂血管旁不规则软组织,多学科会诊后拟诊断为阴道残端病变行腹腔镜探查术,根据术中所见行左侧阴道残端肿物切除术,切除组织经石蜡切片病理检查证实为阴道残端EMs。结合此病例讨论并复习相关文献,为临床上子宫切除术后出现阴道出血病例提供了诊疗思路,旨在提高临床医生对手术后并发阴道残端EMs的认识。
关键词:
Endometriosis (EMs) is a common chronic inflammatory disease in women that refers to the presence of glands and stroma of the endometrium outside the uterine cavity, most commonly in the ovaries, pelvic peritoneal surfaces, or other pelvic tissues. We report a 48-year-old woman who developed vaginal stump endometriosis 15 years after hysterectomy for hysteromyoma. The patient was admitted to the hospital due to irregular vaginal bleeding accompanied by abdominal pain for 3 years and aggravation for 2 months. Enhanced CT of the whole abdomen found irregular soft tissue beside the left iliac vessels. After multidisciplinary consultation, the patient was diagnosed with vaginal stump disease and underwent laparoscopic exploration. Resection of the left vaginal stump mass was performed according to the intraoperative findings, and the excised tissue was confirmed to be EMs by paraffin section pathological examination. Combined with this case, we discussed and reviewed relevant literature to provide diagnosis and treatment ideas for clinical cases of vaginal bleeding after hysterectomy, so as to improve clinicians’ understanding of postoperative vaginal stump EMs.
Keywords:
本文引用格式
陈曦, 胡玉萍, 胡乐, 杨永秀.
CHEN Xi, HU Yu-ping, HU Le, YANG Yong-xiu.
子宫内膜异位症(endometriosis,EMs)是脱落的子宫内膜种植于子宫腔外而引起的一系列临床症状。脱落的子宫内膜主要见于卵巢,还可见于膀胱、肠道、盆腔深部神经、输尿管、腹壁、腹部皮肤,甚至见于膈膜、胸膜、肺、心包和大脑等罕见部位[1]。EMs以雌孕激素依赖为特征[2],症状复杂且不典型,可表现为痛经、性交困难以及与月经周期相关的盆腔痛、下腹痛或腰背痛等,但许多女性也可能无明显症状,或认为是月经期生理反应,甚至认为是胃肠道疾病而错误就诊,患者的不重视及延迟就医导致EMs的诊断通常从症状出现到确诊平均延迟8~12年[3]。子宫切除术后的EMs病例比较罕见,临床上极易误诊、漏诊。现报告1例48岁患者,15年前因子宫肌瘤行子宫切除术,此次因不规则阴道出血伴腹痛加重就诊,术后石蜡切片病理检查证实为EMs。结合此病例并进行文献复习,总结子宫切除术后阴道出血的诊疗思路。
1 病例报告
患者 女,48岁,因不规则阴道出血伴腹痛3年,加重2个月,于2021年3月24日收住兰州大学第一医院(我院)。患者孕2产2,2018年起间断出现不规则阴道出血,约半年至1年1次,每次2~3 d,量少,呈暗红色,有血块;出血期间伴不规律下腹痛及黏液样粪便,腹痛呈慢性钝痛,无放射痛,体位变化后腹痛程度不变,排气、排便后未见明显缓解,否认排便习惯改变,无腹胀,无发热、寒战,未进行治疗。上述症状于2个月前加重,不规则阴道出血频率较前增加,每月1次,每次间断出血约2~3 d,出血量较前稍增多(每次约使用2~3片卫生巾)。近1年患者体质量减轻2 kg。患者已手术绝经15年,2006年因子宫肌瘤而行子宫切除术(未见病历),术后恢复良好,未诉异常。
妇科检查示:外阴发育正常,已婚已产型,阴道畅,黏膜光滑,盆腔空虚,触诊阴道残端略增厚,未触及明显结节,下腹部压痛阳性。经阴道彩色超声示:阴道顶上方、盆腔中央可见大小约2.4 cm×2.2 cm囊实性占位,边界清,回声不均,内可见血流信号,与肠道关系密切,血管阻力指数0.49;右侧卵巢大小2.9 cm×1.8 cm,左侧卵巢未探及异常。全腹增强CT示:①子宫切除术后,术区未见明显异常;②左侧髂血管旁不规则软组织(见图1A),增强可见钙化,考虑转移灶,左侧肾盂和输尿管中、上段扩张积水(见图1B);③直肠下壁段增厚。电子结肠镜见:直肠黏膜散在点状糜烂面,提示直肠炎。人乳头瘤病毒检测为阴性。液基薄层细胞学检查示:无上皮内病变及恶性病变(negative for intraepithelial lesion or malignancy,NILM)。肿瘤标志物检查示:糖类抗原125(carbohydrate antigen 125,CA125)46.7 U/mL,人附睾蛋白4(human epididymis protein 4,HE4)206 pmol/L。
图1
遂进行多学科会诊,会诊意见如下。肛肠外科:该患者此次就诊症状无特异性,且肠镜及直肠指检未见明显异常,故暂不考虑本次就诊病因为肠道原发疾病。泌尿外科:患者盆腔内病变暂无法定性,可行腹腔镜探查。目前有关左侧肾盂、输尿管扩张积水问题泌尿外科主要考虑以下原因:①肿瘤压迫;②炎性组织包裹下段输尿管并机化,造成上端输尿管及肾盂扩张积水。是否保留该侧肾脏,需进一步评估该侧肾功能,若肾功能尚可,待妇科手术后行膀胱镜及输尿管镜检查,并植入输尿管支架。若该侧肾功能差,向家属告知病情后建议切除该侧肾脏。超声科及影像科:影像学见病灶较小,且患者子宫已切除,盆腔空虚,行超声引导穿刺活检时无法避开肠管,风险较大。妇科:妇科检查在窥阴器下未见阴道残端有明显异常结节,故无法经阴道镜取活检。目前主要考虑阴道不规则出血原因主要有:①阴道残端病变:阴道上皮内瘤变?阴道残端癌?②卵巢恶性肿瘤?综合各科室意见后行腹腔镜探查术。
2021年3月30日行腹腔镜探查术,术中见子宫缺如,右侧卵巢与周围组织紧密粘连,难以分辨,左侧卵巢部分与左侧盆腔壁粘连,左侧盆腔壁似冰冻骨盆样,左侧阴道残端增厚;右侧部分肠管、肠系膜与右侧盆腔壁粘连,分解粘连后于右侧附件区可见大小约2 cm×2 cm的包块,表面粗糙,壁厚,囊壁表面可见散在紫蓝色结节,活动度差,与右侧盆腔壁粘连紧密,右侧卵巢及输卵管难以辨认。术中与家属沟通后,其家属要求切除右侧卵巢,保留左侧卵巢。遂行腹腔镜右侧卵巢及输卵管切除术+左侧阴道残端肿物切除术+右侧盆腔淋巴结切除术,切除组织送病理检查。
左侧阴道残端肿物病理检查示:纤维组织内可见异位的子宫内膜腺体及间质,部分腺上皮细胞轻度不典型增生,见图2。免疫组织化学检查示:CD(部分+),Ki-67(20%),p53(-),p16(+),波形蛋白(Vimentin,+),CD10(弱+),雌激素受体(+),孕激素受体(-)。
图2
术后病理诊断:①(右侧卵巢)滤泡囊肿;②(右侧输卵管)充血、水肿;③(左侧阴道残端肿物)慢性炎症伴纤维组织增生;④(阴道残端)结合形态学及免疫组织化学结果,考虑EMs,部分腺上皮细胞轻度不典型增生;⑤(右侧盆腔淋巴结)检出淋巴结6枚,未见明确异形成分。根据术中情况及术后石蜡切片病理结果,最终诊断为:①阴道残端EMs;②子宫切除术后;③直肠炎;④左侧肾积水。
术后予以患者皮下注射醋酸亮丙瑞林微球3.75 mg后转入泌尿外科继续治疗左侧肾积水。2021年4月12日行膀胱镜检查+输尿管镜检查+经尿道输尿管支架植入术+开放左侧输尿管旁肿物切除术+输尿管粘连松解术。术后左侧输尿管旁肿物石蜡切片病理结果提示:①(输尿管肿物)考虑纤维组织增生伴透明变性、钙化及慢性炎症;②(髂血管旁淋巴组织)送检组织均为增生的纤维脂肪组织,纤维组织透明变性,局部可见小片状出血灶。术后随访,现患者恢复较好。
2 讨论
2.1 鉴别诊断
结合本病例,子宫切除术后无明显诱因出现不规则阴道出血症状的患者需考虑以下几种情况。
2.1.1 阴道上皮内瘤变、阴道残端癌
阴道上皮内瘤变是阴道癌的癌前病变,最常见于因宫颈病变而行子宫切除术的病例,2种疾病的主要危险因素有:下生殖道高危型人乳头瘤病毒持续感染、盆腔放疗史、阴道黏膜损伤和吸烟等。高级别阴道上皮内瘤变若不及时治疗,20%的患者会在3年内进展为阴道癌。患者症状不典型,主要表现为阴道流液、下腹痛,妇科检查时可触及痛性结节或包块,与宫颈癌筛查相同,筛查遵从“三阶梯”顺序:细胞学检查、阴道镜检查、活组织病理学检查。此外,磁共振成像(magnetic resonance imaging,MRI)和超声检查等可辅助诊断。
2.1.2 卵巢恶性肿瘤
卵巢癌患者多以腹胀、食欲减退和盆腔包块为临床首发症状[4];部分患者可有阴道流液、近期体质量明显下降等,CA125、甲胎蛋白、HE4升高,妇科彩色超声可见附件区混合性或实性肿物,部分可见乳头状结构,MRI多表现为卵巢实性或囊实性肿块,囊壁结节多发,可见坏死组织,囊壁及分隔增厚,薄厚不均,伴不均匀强化,可同时伴有淋巴结肿大及腹水。
2.1.3 阴道残端Ems
该病多表现为明显的阴道出血、下腹痛,既往多合并深部浸润型EMs,妇科检查时可触及痛性结节或包块,或存在肉眼可见紫蓝色结节,妇科超声可表现为异位病灶周围组织结构欠清晰,回声不均匀,较大的异位病灶可见混合低回声,但诊断的金标准仍然是病理检查。
2.2 病灶形成原因分析
结合文献,本例患者EMs病灶的形成原因考虑有以下几个方面:①患者描述阴道异常出血症状存在一定的周期性,不排除患者既往已有盆腔EMs,而首次手术未彻底清除EMs异位病灶或未将盆腔内脱落的子宫内膜冲洗干净,而当时保留患者卵巢,且卵巢功能旺盛,使得排卵期盆腔暴露于高雌激素状态,而患者术后从未使用药物辅助治疗,过量的雌激素维持着盆腔内残存的异位子宫内膜生长。②首次手术时导致子宫内膜碎片在盆腔残留,有活力的子宫内膜碎片种植到盆腔内,在体内激素影响下经黏附、侵袭形成内盆腔子宫内膜异位病灶。③在首次手术时,脱落的子宫内膜可能经静脉或淋巴管移动到阴道残端、输尿管及肠管周围,受体内激素周期性影响,异位的子宫内膜进一步生长。④根据患者术后病理检查结果,患者盆腔组织存在炎症细胞浸润,结合雌、孕激素作用,促进已有EMs病灶的发展,或直接参与盆腔EMs病灶形成,并在输尿管周围形成炎性浸润,逐步纤维化,进一步引起肾盂和输尿管中、上段扩张积水。⑤有研究认为出现EMs临床症状的女性可能具有EMs遗传易感性,EMs患者的体腔上皮(腹膜)、子宫内膜、子宫内膜脉管系统和其他盆腔组织中存在祖细胞/干细胞的表观基因组异常,具有正常功能的子宫内膜上皮细胞包含许多突变,其中一些突变广泛存在于EMs患者的子宫内膜上皮细胞中,例如KRAS、PIK3CA和ARID1A基因,这些突变基因可能在异位内膜碎片的种植和生长中发挥作用[2]。
总之,阴道残端EMs少见,在国内外均为个案报道,因症状不典型,临床中容易误诊或漏诊,进而延误治疗。因此,子宫切除术后出现异常阴道出血、腹痛及输尿管积水等症状时,诊断时需将阴道残端病变与阴道残端EMs相鉴别。此外,行全子宫切除术时,一定要仔细探查整个盆腹腔,如果术中已经发现EMs病灶,需尽量切净,且术后给予药物治疗,以降低复发率。手术操作时,使用生理盐水冲洗盆腹腔并注意保护皮肤切口,取出子宫的过程中尽量避免子宫内膜组织接触阴道残端,并彻底消毒阴道残端。
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