孕期MRI对Galen静脉动脉瘤样畸形胎儿新生儿期高危的预测价值

Value of MRI in fetuses with vein of Galen aneurysmal malformation to the prediction of high risks in neonatal period

目的探讨MRI定量参数对Galen静脉动脉瘤样畸形(vein of Galen aneurysmal malformation,VGAM)胎儿新生儿期高危的预测价值。方法孕晚期经MRI检查诊断胎儿VGAM的单胎妊娠孕妇13例,由2名高年资医师采用双盲法测量胎儿直窦或永存镰状窦最小上下径及对应层面最大左右径,并计算直窦或永存镰状窦面积。13例经医学咨询后均选择继续妊娠,出生后行头颅、心脏超声随诊,4例病情轻微于婴儿期择期行血管内栓塞治疗者为婴儿组,9例新生儿期死亡或紧急行血管内栓塞治疗者为新生儿高危组。比较2组胎儿直窦或永存镰状窦最小上下径及对应层面最大左右径、面积;采用组内相关系数(intraclass correlation efficient,ICC)评估2名医师测量结果的一致性;绘制ROC曲线,评估直窦或永存镰状窦最小上下径及对应层面最大左右径、面积预测VGAM胎儿新生儿期高危的效能。结果A、B医师测量的新生儿高危组直窦或永存镰状窦最大左右径[(8.87±1.88)、(8.81±1.89)mm]均长于婴儿组[(5.98±0.83)、(5.93±0.81)mm](t=2.904,P=0.014;t=2.885,P=0.015),面积[(74.24±24.88)、(73.49±24.94)mm^(2)]均大于婴儿组[(49.44±8.56)、(47.96±8.02)mm^(2)](t=2.659,P=0.023;t=2.766,P=0.019),最小上下径[(8.34±2.01)、(8.31±2.04)mm]与婴儿组[(8.25±0.56)、(8.08±0.64)mm]比较差异均无统计学意义(t=0.123,P=0.905;t=0.303,P=0.768)。A、B医师测量的直窦或永存镰状窦最小上下径及对应层面最大左右径、面积的ICC分别为0.996(95%CI:0.985~0.999)、1.000(95%CI:0.958~1.000)、0.998(95%CI:0.985~1.000),一致性较好。胎儿直窦或永存镰状窦最大左右径、面积分别以6.86mm、59.00mm^(2)为最佳截断值,预测VGAM胎儿新生儿期高危的AUC分别为0.972(95%CI:0.890~1.000,P=0.009)、0.806(95%CI:0.562~1.000,P=0.047),灵敏度分别为89%、67%,特异度均为100%。结论直窦或永存镰状窦最小上下径对应层面最大左右径及面积增大的VGAM胎儿新生儿期高危风险增大,最大左右径在预测VGAM胎儿新生儿期高危中有较高价值。

Objective To invesitage the value of MRI quantitative parameters of fetuses with vein of Galen aneurysmal malformation(VGAM)to the prediction of high risks in neonatal period.Methods Thirteen singleton pregnant women with MRI diagnosed fetal VGAM in the third trimester were measured the minimum superior-to-inferior(SI)diameter of the straight sinus/persistent falciform sinus and the maximum right-to-left(RL)diameter of the corresponding layer by two senior physicians in a double-blind method,and the area of the straight sinus/persistent falciform sinus was calculated.All 13cases chose to continue their pregnancy after medical consultation,and underwent cranial and cardiac ultrasound follow-up after delivery.Four infants with mild VGAM selected endovascular embolization(infantile group),and the other 9neonates with severe symptoms died in the neonatal period or underwent emergency endovascular embolization(neonatal high risk group).The minimum SI diameter of straight sinus/persistent falciform sinus,the maximum RL diameter of the corresponding layer and the area were compared between two groups.The intraclass correlation efficient(ICC)was used to evaluate the consistency of the measurement results between two physicians,and the ROC curves were drawn to evaluate the efficiencies of the minimum SI diameter of straight sinus/persistent falciform sinus,maximum RL diameter of the corresponding layer and area on predicting the high risks in neonatal period.Results The maximum RL diameters of straight sinus/persistent falciform sinus measured by physicians A and B were longer in neonatal high risk group[(8.87±1.88),(8.81±1.89)mm]than those in infantile group[(5.98±0.83),(5.93±0.81)mm](t=2.904,P=0.014;t=2.885,P=0.015),the areas were larger in neonatal high risk group[(74.24±24.88),(73.49±24.94)mm^(2)]than those in infantile group[(49.44±8.56),(47.96±8.02)mm^(2)](t=2.659,P=0.023;t=2.766,P=0.019),and the minimum SI diameters showed no significant differences between neonatal high risk group[(8.34±2.01),(8.31±2.04)mm]and infantile group[(8.25±0.56),(8.08±0.64)mm](t=0.123,P=0.905;t=0.303,P=0.768).The ICCs of mininum SI diameter,maximum RL diameter,and area measured by physicians A and B were 0.996(95%CI:0.985-0.999),1.000(95%CI:0.958-1.000)and 0.998(95%CI:0.985-1.000),respectively,indicating a high consistency.When the optimal cut-off values of maximum RL diameter and area were 6.86mm and 59.00mm^(2),the AUCs for predicting neonatal high risks were 0.972(95%CI:0.890-1.000,P=0.009)and 0.806(95%CI:0.562-1.000,P=0.047),the sensitivities were 89%and 67%,and the specificities were 100%and 100%,respectively.Conclusion The VGAM fetuses with increased minimum SI diameter of straight sinus/persistent falciform sinus,maximum RL diameter of the corresponding layer and area are at a high risk in the neonatal period,and the maximum RL diameter has a high value to the prediction of high risks in neonatal period of VGAM fetuses.