妊娠合并腹膜播散性平滑肌瘤病一例并文献复习

Leiomyomatosis peritonealis disseminata during pregnancy: a case report and literature review

目的研究妊娠合并腹膜播散性平滑肌瘤病的临床特点。方法报告青岛市胶州中心医院收治的1例妊娠合并腹膜播散性平滑肌瘤病诊疗经过。检索国内外妊娠合并腹膜播散性平滑肌瘤病的相关文献,分析腹膜播散性平滑肌瘤病的临床特点及母儿预后等资料。结果(1)病例报告:患者33岁,孕37周^(+2)时因“发现盆腔包块7个月,阴道流液半小时”入院。入院当日行子宫下段剖宫产+盆腹腔肿物切除术。术后随访26个月,未见明显复发迹象。(2)文献复习:检索到相关文献13篇,共15例(含本例)。15例患者发病年龄32岁(19~47岁),其中5例有肌瘤切除史。12/15孕期无明显症状,于分娩时或产后发现;3/15孕期出现明显症状,以腹痛为主。6/15发生妊娠期高血压疾病、胎膜早破、胎位异常、羊水过少等产科相关并发症。2/15例为辅助生殖技术妊娠。11/15分娩孕周>32周,3/15分娩孕周<28周,1/15因严重并发症于孕10周终止妊娠。12/15采取剖宫产术+肌瘤清除术,并保留子宫和附件;3/15例采取了子宫切除术(其中2例同时切除双侧附件)。7例病理表现为广泛、大量分布的小肌瘤,类似恶性肿瘤浸润,直径0.5~1.0 cm;5例有多个较大肌瘤,伴随散在分布的小肌瘤。结论妊娠合并腹膜播散性平滑肌瘤病是一种较罕见的良性疾病,常缺乏明显的临床症状,术前较难诊断,多于术后经组织病理学确诊,可能误诊为恶性肿瘤。通常选择剖宫产术+肌瘤切除术治疗。

Objective To review the clinical and pathological features,pathogenesis,and differential diagnosis of leiomyomatosis peritonealis disseminata(LPD)during pregnancy.Methods We report the diagnosis and treatment of LPD in a pregnant woman admitted to Jiaozhou Central Hospital of Qingdao.We retrieved relevant literature from China and abroad to review the clinical characteristics and outcomes.Results(1)Case report:The 33-year-old pregnant woman who had had previous cesarean sections was admitted at 37^(+2) weeks gestation due to premature rupture of membranes 30 minutes before arriving at the hospital with a pelvic mass which was first diagnosed seven months earlier,in the first trimester.A lower segment cesarean section was performed,and the pelvic mass was removed on admission.No relapse was reported during the postoperative follow-up for 26 months.(2)Literature review:thirteen papers involving 15 cases,including the present case,were retrieved.The age at onset was 32 years old(19-47 years old);five of them had a history of myomectomy.Twelve cases had no significant symptoms,and the diagnosis was made at delivery or postpartum;the other three had noticeable features,mainly with abdominal pain.Six had obstetrical complications,including hypertensive disorders of pregnancy,premature rupture of membrane,abnormal fetal position,and oligohydramnios.Two cases had a history of assisted reproductive technology.Eleven cases delivered after 32 gestational weeks,three before 28 weeks,one aborted at ten gestational weeks due to severe complications.Twelve had cesarean section and myomectomy and the uterus and appendages were preserved.Three cases had a cesarean hysterectomy and two of them also had bilateral salpingo-oophorectomy.The pathological manifestations of seven cases were extensive and massively distributed small fibroids,similar to malignant tumor infiltration,with the diameter of 0.5 to 1.0 cm;five cases had multiple large fibroids,accompanied by scattered small fibroids.Conclusion LPD during pregnancy is rare,and usually benign,with no obvious clinical symptoms.LPD is more likely to be misdiagnosed as a malignant tumor before the operation,and the diagnosis is usually confirmed by postoperative histopathology.A common surgical strategy is a cesarean section combined with pelvic mass resection.