Kunpalin Y, Sahakyan Y, Sander B, Snelgrove J W, Raghuram K, Kulkarni A V, Van Mieghem T
Fetal Medicine Unit, Department of Obstetrics & Gynaecology, Mount Sinai Hospital and University of Toronto, Toronto, ON, Canada.
Toronto Health Economics and Technology Assessment Collaborative, Toronto General Hospital Research Institute, University Health Network, Toronto, ON, Canada.
Ultrasound Obstet Gynecol. 2025 Jul 31. doi: 10.1002/uog.29289.
Currently, there are several surgical approaches to manage fetal open spina bifida (OSB), namely postnatal surgical repair, open fetal surgery and its minimally invasive alternative, fetoscopic repair. Our objective was to determine the optimal surgical approach for OSB, weighing the benefits and risks to the fetus and the pregnant woman.
We assessed the health outcomes of open fetal, fetoscopic and postnatal surgical repair for pregnant women (mean ± SD age, 31 ± 3 years) with a singleton pregnancy and their offspring with OSB using a decision analytic model. We projected expected quality-adjusted life years (QALYs) associated with each of the interventions, discounted at 1.5% annually over the lifetime time horizon for pregnant women and their offspring. Secondary maternal outcomes during the pregnancy included delivery mode and complications such as chorioamnionitis, uterine dehiscence, placental abruption, pulmonary embolism and death. Offspring outcomes included preterm birth, perinatal and postnatal mortality, cerebrospinal fluid (CSF) diversion surgery by 12 months of age and wheelchair use at 30 months of age. Our model was populated using data from the published literature and by consultation with clinical experts. Deterministic and probabilistic sensitivity analyses were conducted.
Fetoscopic and open fetal surgery resulted in an identical number of expected QALYs (38.02 per mother-offspring dyad) and translated into a QALY gain of 1.70 per dyad compared with postnatal repair. With respect to QALYs gained, the probabilistic analyses showed that fetoscopic surgery was the preferred strategy in 51% of simulations, and open fetal surgery in the remaining 49% of simulations. When compared with postnatal repair, both open fetal and fetoscopic surgery showed that the gains in QALYs were most sensitive to the disutility associated with CSF diversion surgery and to the rate of wheelchair use. When comparing open fetal and fetoscopic approaches, the results were highly sensitive to the accuracy of all treatment effect estimates.
In the management of fetal OSB, both fetoscopic and open fetal surgery demonstrate superior QALY gains compared with postnatal repair, largely related to a reduced number of individuals who use a wheelchair or require CSF diversion surgery. Given similar effectiveness of fetoscopic and open fetal surgery, an individual risk assessment is essential to guide decision-making between these two surgical approaches. © 2025 The Author(s). Ultrasound in Obstetrics & Gynecology published by John Wiley & Sons Ltd on behalf of International Society of Ultrasound in Obstetrics and Gynecology.
目前，有几种手术方法可用于治疗胎儿开放性脊柱裂（OSB），即产后手术修复、开放性胎儿手术及其微创替代方法——胎儿镜修复。我们的目的是确定治疗OSB的最佳手术方法，权衡对胎儿和孕妇的益处与风险。
我们使用决策分析模型评估了单胎妊娠且胎儿患有OSB的孕妇（平均年龄±标准差，31±3岁）及其后代接受开放性胎儿手术、胎儿镜手术和产后手术修复后的健康结局。我们预测了与每种干预措施相关的预期质量调整生命年（QALY），并在孕妇及其后代的终身时间范围内按每年1.5%进行贴现。孕期的次要母亲结局包括分娩方式和并发症，如绒毛膜羊膜炎、子宫裂开、胎盘早剥、肺栓塞和死亡。后代结局包括早产、围产期和产后死亡率、12个月龄前进行脑脊液（CSF）分流手术以及30个月龄时使用轮椅的情况。我们的模型使用已发表文献中的数据并通过咨询临床专家来构建。进行了确定性和概率敏感性分析。
胎儿镜手术和开放性胎儿手术导致的预期QALY数量相同（每对母婴为38.02），与产后修复相比，每对母婴的QALY增益为1.70。就获得的QALY而言，概率分析表明，在51%的模拟中胎儿镜手术是首选策略，在其余49%的模拟中开放性胎儿手术是首选策略。与产后修复相比，开放性胎儿手术和胎儿镜手术均表明，QALY的增益对与CSF分流手术相关的负效用以及轮椅使用率最为敏感。在比较开放性胎儿手术和胎儿镜手术方法时，结果对所有治疗效果估计的准确性高度敏感。
在胎儿OSB的治疗中，与产后修复相比，胎儿镜手术和开放性胎儿手术均显示出更高的QALY增益，这在很大程度上与使用轮椅或需要CSF分流手术的个体数量减少有关。鉴于胎儿镜手术和开放性胎儿手术效果相似，个体风险评估对于指导这两种手术方法之间的决策至关重要。© 2025作者。《超声妇产科》由约翰·威利父子有限公司代表国际妇产科超声学会出版。
