Endoscopic surgery for the antenatal treatment of myelomeningocele: the CECAM trial
Endoscopic surgery for the antenatal treatment of myelomeningocele: the CECAM trial
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DOI：
10.1016/j.ajog.2015.09.065
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发表时间：
2016-01-01
影响因子：
9.8
通讯作者：
Quintero, Ruben A.
中科院分区：
文献类型：
--
作者：
Pedreira, Denise A. L.;Zanon, Nelci;Quintero, Ruben A.
背景技术背景：最近一项名为脊髓脊膜膨出治疗研究的随机临床试验（MOMS试验）表明，通过开放性胎儿手术进行开放性脊柱裂（OSB）的产前矫正与出生后修复相比，婴儿神经功能结局改善相关，但以增加孕产妇发病率为代价。目的：我们试图报告我们第一阶段试验的最终结果（Cirurgia Endoscopica帕拉Correcao Ceramiatalda Meningomielocele [CECAM]）的可行性、安全性、潜在受益，和副作用的胎儿镜治疗OSB使用我们独特的手术technique.Study设计：10个连续妊娠腰骶部OSB参加了这项研究。手术均在全麻下经皮穿刺进行，采用3个端口和部分二氧化碳吹入。在对胎儿进行适当的手术定位后，用剪刀释放神经基板，并破坏皮肤，在病变处放置生物纤维素贴片。使用单针连续缝合将皮肤缝合在贴片上。术前，术后和出生后进行磁共振成像，以评估后脑疝。在出院前和3、6和12个月时进行神经发育评估。所有病例均通过剖宫产分娩，此时评估子宫变薄或裂开的证据。结果：手术时的中位孕龄为27周（范围25-28周）。10只胎儿中有8只完成了内镜修复。2例因子宫入路丢失而失败。出生时的平均胎龄为32.4周，手术和分娩之间的平均潜伏期为5.6周（范围2-8周）。1例胎儿和1例新生儿死亡，1例不成功的病例进行了产后修复。在7例可供分析的婴儿中，6例发生了后脑疝完全逆转。3名婴儿需要脑室腹腔分流术或第三脑室造口术。7例中6例功能性运动水平与解剖水平相同或更好。没有明显的产妇发病率，也没有子宫肌层变薄或裂开的证据。然而，手术复杂的胎膜早破和premature.CONCLUSION：我们的研究表明，产前治疗OSB使用胎儿镜的方法和我们独特的手术技术可以导致水密密封，逆转后脑疝，和比预期更好的运动功能。我们的技术与先前开放式胎儿手术和胎儿镜研究中使用的OSB经典修复方法有很大不同，后者切开硬脑膜，多层闭合缺损。相反，我们使用生物纤维素贴片放置在病变和简单的皮肤关闭。因此，我们的技术是一种替代目前的模式，在产前治疗的OSB。我们的临床结果与我们之前广泛的动物研究结果一致。我们的方法和技术的产妇的好处包括最低的发病率和没有子宫肌层的遗产。目前该方法的局限性包括潜在的通路丢失、胎膜早破和伴随的早产。需要进行II期试验来预防这些并发症，并进一步评估我们独特的手术方法和技术的风险和益处。
BACKGROUND: A recent randomized clinical trial named Management of Myelomeningocele Study (MOMS trial) showed that prenatal correction of open spina bifida (OSB) via open fetal surgery was associated with improved infant neurological outcomes relative to postnatal repair, but at the expense of increased maternal morbidity.OBJECTIVE: We sought to report the final results of our phase I trial (Cirurgia Endoscopica para Correcao Antenatalda Meningomielocele [CECAM]) on the feasibility, safety, potential benefits, and side effects of the fetoscopic treatment of OSB using our unique surgical technique.STUDY DESIGN: Ten consecutive pregnancies with lumbosacral OSB were enrolled in the study. Surgeries were performed percutaneously under general anesthesia with 3 ports and partial carbon dioxide insufflation. After appropriate surgical positioning of the fetus, the neuro-placode was released with scissors and the skin was undermined to place a biocellulose patch over the lesion. The skin was closed over the patch using a single running stitch. Preoperative, postoperative, and postnatal magnetic resonance imaging were performed to assess hindbrain herniation. Neurodevelopmental evaluation was performed before discharge and at 3, 6, and 12 months. All cases were delivered by cesarean delivery, at which time the uterus was assessed for evidence of thinning or dehiscence.RESULTS: The median gestational age at the time of surgery was 27 weeks (range 25-28 weeks). Endoscopic repair was completed in 8 of 10 fetuses. Two cases were unsuccessful due to loss of uterine access. The mean gestational age at birth was 32.4 weeks with a mean latency of 5.6 weeks between surgery and delivery (range 2-8 weeks). There was 1 fetal and 1 neonatal demise, and 1 unsuccessful case underwent postnatal repair. Of the 7 infants available for analysis, complete reversal of hindbrain herniation occurred in 6 of 7 babies. Three babies required ventriculoperitoneal shunting or third ventriculostomy. Functional motor level was the same or better than the anatomical level in 6 of 7 cases. There was no significant maternal morbidity and no evidence of myometrial thinning or dehiscence. However, surgeries were complicated by premature rupture of membrane and prematurity.CONCLUSION: Our study suggests that the antenatal treatment of OSB using a fetoscopic approach and our unique surgical technique can result in a watertight seal, reversal of the hindbrain herniation, and better than expected motor function. Our technique differs substantially from the classic repair of OSB used in prior open fetal surgery and fetoscopic studies, in which the dura mater is dissected and the defect is closed in multiple layers. Instead, we use a biocellulose patch placed over the lesion and simple closure of the skin. As such, our technique is an alternative to the current paradigms in the antenatal treatment of OSB. Our clinical outcomes are in line with the results of our extensive prior animal work. Maternal benefits of our approach and technique include minimal morbidity and no myometrial legacy. Current limitations of the approach include potential loss of access, premature rupture of membranes, and attendant prematurity. Phase II trials are needed to prevent these complications and to further assess the risks and benefits of our distinct surgical approach and technique.