Uterine Defect after Open Maternal-fetal Surgery

Abstract

A 36-year-old gravida 5, para 3 woman presented with persistent dysmenorrhea, dyspareunia, and abnormal uterine bleeding (AUB) for 4 years following an open maternal-fetal surgery (OMFS) for fetal myelomeningocele repair. Surgery was performed at 23-week gestation and involved a full-thickness hysterotomy at the posterofundal aspect of the uterus, followed by placement of an omental patch over the hysterotomy site. A cesarean section was ultimately performed at 31-week gestation because of preterm labor. Attempts at uterine exteriorization were unsuccessful because of omental adhesions. The postpartum course was uncomplicated. In the workup of her gynecologic symptoms, a transvaginal ultrasound was performed. A fundal defect in the uterine myometrium was seen [Fig. 1]. The patient ultimately underwent a total laparoscopic hysterectomy, lysis of omental adhesions [Fig. 2], and bilateral salpingectomy after failing medical management. A fundal uterine defect was again noted in the pathologic assessment [Fig. 3]. The postoperative course was uncomplicated, and her pelvic pain resolved.Fig. 2(A) Uterus with omental adhesions to posterior fundus. (B) Otherwise normal appearing posterior cul-de-sac.View Large Image Figure ViewerDownload Hi-res image Download (PPT)Fig. 3Fundal uterine defect noted during pathologic assessment following total laparoscopic hysterectomy. Omentum was adhered to the uterine serosa overlying the defect. C = cervix E = endometrium and cavity; M = myometrium.View Large Image Figure ViewerDownload Hi-res image Download (PPT) This case highlights potential gynecologic complications following OMFS that may be overlooked. New onset gynecologic symptoms including chronic abdominal/pelvic pain, and AUB have been reported following both OMFS [1Wilson RD Lemerand K Johnson MP et al.Reproductive outcomes in subsequent pregnancies after a pregnancy complicated by open maternal-fetal surgery (1996–2007).Am J Obstet Gynecol. 2010; 203: 209.e1-209.e6Abstract Full Text Full Text PDF Scopus (82) Google Scholar] and fetoscopic surgery [2Sacco A Van der Veeken L Bagshaw E et al.Maternal complications following open and fetoscopic fetal surgery: a systematic review and meta-analysis.Prenat Diagn. 2019; 39: 251-268Crossref PubMed Scopus (49) Google Scholar]. Neither OMFS nor fetoscopic surgery has been associated with infertility [1Wilson RD Lemerand K Johnson MP et al.Reproductive outcomes in subsequent pregnancies after a pregnancy complicated by open maternal-fetal surgery (1996–2007).Am J Obstet Gynecol. 2010; 203: 209.e1-209.e6Abstract Full Text Full Text PDF Scopus (82) Google Scholar,2Sacco A Van der Veeken L Bagshaw E et al.Maternal complications following open and fetoscopic fetal surgery: a systematic review and meta-analysis.Prenat Diagn. 2019; 39: 251-268Crossref PubMed Scopus (49) Google Scholar]. In this case, new onset of pelvic pain, dysmenorrhea, and AUB were associated with a posterofundal uterine defect, complicated by prior OMFS. These symptoms mimic those that can arise following cesarean sections, complicated by postoperative cesarean scar isthmocele formation [3Morris H Surgical pathology of the lower uterine segment caesarean section scar: is the scar a source of clinical symptoms?.Int J Gynecol Pathol. 1995; 14: 16-20Crossref PubMed Scopus (135) Google Scholar]. Symptom etiology in the setting of an isthmocele may be related to underlying lymphocytic infiltration, abnormal vascularity, and iatrogenic adenomyosis [3Morris H Surgical pathology of the lower uterine segment caesarean section scar: is the scar a source of clinical symptoms?.Int J Gynecol Pathol. 1995; 14: 16-20Crossref PubMed Scopus (135) Google Scholar]. This case raises the question as to whether a similar underlying pathophysiologic process accounts for the symptoms, as focal adenomyosis was found around the defect but lymphocytic infiltration or abnormal vascularity was not distinctly commented on. With the increasing prevalence of intrauterine maternal-fetal surgery, more long-term follow-up studies are required to better understand both the reproductive outcomes and gynecologic implications of these procedures.