目的:选择开放性脊柱裂(OSB)的胎儿进行胎儿手术,以改善长期预后。使用先进的磁共振成像(MRI)定量的体积,表面积,大脑结构的形状,和使用对应于回转的参数的表面曲率分析,我们研究了胎儿手术对大脑发育的影响。
方法:我们比较了29个OSB胎儿在胎儿手术前(<24周)的MRI图像,手术后一周和六周,至36个年龄匹配的正常对照(21+2-36+2周)。自动超分辨率重建提供了3D各向同性体积脑图像。无髓鞘白质,小脑和脑室被自动分割和手动细化;脑容量,表面积,和形状参数(体积/表面积)被定量。使用数学标记(形状指数(SI)和曲率)来测量回转性。根据病变类型评估参数(例如,骨髓分裂(MS)),术后持续性后脑疝(HH),以及存在幕上异常,即call体部分发育不全(pACC),和异位症(HT)。
结果:手术后六周,OSB心室容积增长(mm3/周)和形状参数(mm/周)高于对照组;(2500.94,IQR:1665.51-3604.1mm3/周vs708.21,IQR:435.06-965.77mm3/周,p<0.001),和(0.075,IQR:0.047-0.112毫米/周vs0.022,IQR:0.009-0.042毫米/周,p=0.046)。在pACC(p<0.001)和持续性HH(p=0.002)的情况下,心室容积增长在手术后六周增加。在此期间,与对照组相比,OSB无髓鞘白质形状参数变化(毫米/周)降低(0.056,IQR:0.044-0.092毫米/周vs0.159,IQR:0.1-0.247毫米/周,p=0.002),特别是在持续性HH(p=0.011)的情况下,MS(p=0.015),HT(p=0.022),具有CC异常的HT(p=0.017),和持续性HH,CC异常(p=0.007)。尽管曲率/周(mm-1)显着降低,但术后6周(0.061,IQR:0.040-0.093mm-1/周vs0.094,IQR:0.072-0.145mm-1/周,p<0.001)与一周后的初始增加相比(0.144,IQR:0.101-0.233mm-1/周vs0.072,IQR:0.059-0.08mm-1/周,p<0.001),与对照组相比,使用SI的旋化总体上有所增加。这一观察结果在患有pACC的胎儿中更为突出,和严重的脑室增宽(p值范围=0.006至<0.001)。
结论:胎儿OSB修复后,volume,心室和无髓鞘白质的形状和弯曲度与正常胎儿有显著差异.胎儿手术后的脑形态变化不仅限于对CSF循环的影响。这些观察结果可能对产后结局具有神经认知意义。本文受版权保护。保留所有权利。
Prenatal surgery is offered for selected fetuses with open spina bifida (OSB) to improve long-term outcome. We studied the effect of fetal OSB surgery on brain development using advanced magnetic resonance imaging (MRI) techniques to quantify the volume, surface area and shape of cerebral structures and to analyze surface curvature by means of parameters that correspond to gyrification.
We compared MRI data from 29 fetuses with OSB before fetal surgery (mean gestational age (GA), 23 + 3 weeks) and at 1 and 6 weeks after surgery, with that of 36 GA-matched control fetuses (GA range, 21 + 2 to 36 + 2 weeks). Automated super-resolution reconstruction provided three-dimensional isotropic volumetric brain images. Unmyelinated white matter, cerebellum and ventricles were segmented automatically and refined manually, after which volume, surface area and shape parameter (volume/surface area) were quantified. Mathematical markers (shape index (SI) and curvedness) were used to measure gyrification. Parameters were assessed according to lesion type (myelomeningocele vs myeloschisis (MS)), postoperative persistence of hindbrain herniation (HH) and the presence of supratentorial anomalies, namely partial agenesis of the corpus callosum (pACC) and heterotopia (HT).
Growth in ventricular volume per week and change in shape parameter per week were higher at 6 weeks after surgery in fetuses with OSB compared with controls (median, 2500.94 (interquartile range (IQR), 1689.70-3580.80) mm3 /week vs 708.21 (IQR, 474.50-925.00) mm3 /week; P < 0.001 and 0.075 (IQR, 0.047-0.112) mm/week vs 0.022 (IQR, 0.009-0.042) mm/week; P = 0.046, respectively). Ventricular volume growth increased 6 weeks after surgery in cases with pACC (P < 0.001) and those with persistent HH (P = 0.002). During that time period, the change in unmyelinated white-matter shape parameter per week was decreased in OSB fetuses compared with controls (0.056 (IQR, 0.044-0.092) mm/week vs 0.159 (IQR, 0.100-0.247) mm/week; P = 0.002), particularly in cases with persistent HH (P = 0.011), MS (P = 0.015), HT (P = 0.022), HT with corpus callosum anomaly (P = 0.017) and persistent HH with corpus callosum anomaly (P = 0.007). At 6 weeks postoperatively, despite OSB fetuses having a lower rate of change in curvedness compared with controls (0.061 (IQR, 0.040-0.093) mm-1 /week vs 0.094 (IQR, 0.070-0.146) mm-1 /week; P < 0.001), reversing the trend seen at 1 week after surgery (0.144 (IQR, 0.099-0.236) mm-1 /week vs 0.072 (IQR, 0.059-0.081) mm-1 /week; P < 0.001), gyrification, as determined using SI, appeared to be increased in OSB fetuses overall compared with controls. This observation was more prominent in fetuses with pACC and those with severe ventriculomegaly (P-value range, < 0.001 to 0.006).
Following fetal OSB repair, volume, shape and curvedness of ventricles and unmyelinated white matter differed significantly compared with those of normal fetuses. Morphological brain changes after fetal surgery were not limited to effects on the circulation of cerebrospinal fluid. These observations may have implications for postnatal neurocognitive outcome. © 2023 The Authors. Ultrasound in Obstetrics & Gynecology published by John Wiley & Sons Ltd on behalf of International Society of Ultrasound in Obstetrics and Gynecology.