BACKGROUND:Children with congenital heart disease are at risk for impaired neurodevelopment (ND). We investigated the association of fetal cerebrovascular resistance with ND in patients with single ventricle lesions. METHODS:In the Single Ventricle Reconstruction (SVR) and Infant Single Ventricle trials, 14-month ND was assessed using the Bayley Scales of Infant Development II. We investigated associations between ND scores and fetal middle cerebral artery pulsatility index (MCA-PI) z-scores, a Doppler-derived estimate of cerebrovascular resistance in a subset of those infants. RESULTS:Neurodevelopment assessments were performed at age 14.3 ± 1 months in 170 (74%) of 230 Infant Single Ventricle and 321 (58%) of 555 SVR subjects. Fetal echocardiographic data were available in 119 subjects, 72 (61%) of which had ND testing. Mean Psychomotor Development Index (PDI) (76 ± 20) and Mental Development Index (MDI) (89 ± 17) scores were lower than normative means (100 ± 15, P < .001). Mean MCA-PI z-score was -0.95 ± 1.52. Middle cerebral artery pulsatility index z-score correlated negatively with PDI (r = -0.27, P = .02) but was not associated with MDI. When MCA-PI z-score was added to a multivariable model controlling for factors identified in the SVR trial to predict PDI, the percentage of explained variation increased from 23% to 30%, and MCA-PI z-score remained an independent predictor (r = -3.864, P = .03). Middle cerebral artery pulsatility index z-score was not an independent predictor in a model adjusting for site. CONCLUSIONS:Among fetuses with single ventricle anomalies, lower cerebrovascular resistance was associated with higher ND scores. This relationship is opposite to that observed with advanced intrauterine growth retardation and may represent a unique ability of these congenital heart disease fetuses to compensate for diminished cerebral oxygen delivery.

译文

背景:患有先天性心脏病的儿童有神经发育受损(ND)的风险。我们调查了单脑室病变患者的胎儿脑血管阻力与ND的关系。
方法:在单室重构(SVR)和婴儿单室试验中,使用贝利婴儿发育量表II评估14个月的ND。我们调查了ND评分与胎儿中脑动脉搏动指数(MCA-PI)z评分之间的关​​联,该评分是多普勒推论得出的这些婴儿子集中的脑血管抵抗力的估计值。
结果:在230名婴儿单心室中的170名(74%)和555名SVR受试者中的321名(58%)进行了14.3±1个月的神经发育评估。 119位受试者可获得胎儿超声心动图数据,其中72位(61%)接受了ND测试。平均精神运动发育指数(PDI)(76±20)和精神发育指数(MDI)(89±17)得分低于标准均值(100±15,P <.001)。平均MCA-PI z评分为-0.95±1.52。大脑中动脉搏动指数z评分与PDI呈负相关(r = -0.27,P = .02),但与MDI不相关。当将MCA-PI z分数添加到控制SVR试验中确定的预测PDI的因素的多变量模型中时,解释的差异百分比从23%增加到30%,而MCA-PI z分数仍是独立的预测因子( r = -3.864,P = .03)。在调整部位的模型中,大脑中动脉搏动指数z评分不是独立的预测因子。
结论:在具有单心室异常的胎儿中,较低的脑血管阻力与较高的ND评分有关。这种关系与子宫内晚期发育迟缓所观察到的相反,并且可能代表了这些先天性心脏病胎儿弥补脑氧输送减少的独特能力。

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