目的:尽管许多研究支持经胎盘治疗胎儿室上性心律失常的疗效,产前抗心律失常治疗后的长期神经发育结局尚不清楚.这项研究的目的是调查矫正年龄36个月时的预后和神经发育结局以及出生后快速性心律失常的发生率。遵循协议定义的产前治疗胎儿室上性快速性心律失常。
方法:这是一项多中心试验的3年随访研究,该试验评估了方案定义的经胎盘治疗胎儿室上性心动过速(SVT)和房扑(AFL)的疗效和安全性。主要终点是校正年龄36个月时的死亡率和神经发育障碍(NDI)。NDI被定义为以下任何一种结果:脑瘫,双侧失明,双侧耳聋或神经发育迟缓。使用适当的发育商量表评估神经发育迟缓,主要是京都心理发展量表,或儿科神经科医生的检查。还评估了出生时以及矫正年龄18和36个月时的快速性心律失常的检出率作为次要终点。此外,我们分析了36个月时NDI与围产期和产后因素的相关性.
结果:在原始试验中招募的50名患者中,一个撤回同意,两个有胎儿死亡,在这项随访研究中,有47例患者可供纳入。其中,45例患儿失访后可进行分析。中位随访时间为3.2,死亡率为2.2%(1/45)(范围,2.1-9.4)年。婴儿在2.1岁时死亡。另一个婴儿缺少神经发育评估数据。在剩下的43名婴儿中,在矫正年龄36个月时,总体上在9.3%(4/43)和三分之二(66.7%)的胎儿水肿伴皮下水肿的病例中检测到NDI。在两名胎龄早期出现严重皮下水肿或腹水的婴儿中发现了脑瘫。在两名患有严重先天性异常(一名患有结节性硬化症,另一名患有异位综合征)的婴儿中发现了神经发育迟缓。在新生儿期,快速性心律失常的发生率为31.9%(15/47),在矫正年龄18和36个月时下降至8.9%(4/45)和4.5%(2/44)。分别。与没有NDI的婴儿相比,诊断时的中位心室率明显更高(265对229bpm;P=0.003)。在患有NDI的婴儿中,与没有的相比,诊断时皮下水肿的胎儿水肿更为常见(50.0%vs2.6%;P=0.019),胎儿积液的持续时间更长(中位数,10.5天vs0天;P=0.013)。出生后心律失常和身体发育异常与NDI无关。
结论:这项为期3年的多中心随访研究首次证明了在方案定义的经胎盘治疗胎儿SVT和AFL后出生的婴儿的长期死亡率和发病率。NDI与胎儿水肿的存在相关,诊断时皮下水肿和胎儿积液持续时间较长。仅在患有严重先天性异常的婴儿中检测到神经发育迟缓。因此,在经过产前治疗胎儿快速性心律失常且没有合并症的婴儿中,NDI的风险很低。然而,在那些有皮下水肿和/或相关的严重先天性异常的胎儿水肿,长期神经系统疾病的发病风险可能会有所增加.©2022国际妇产科超声学会。
Although many studies have supported the efficacy of transplacental treatment for fetal supraventricular tachyarrhythmia, the long-term neurodevelopmental outcome after antenatal antiarrhythmic treatment is not well understood. The aim of this
study was to investigate the prognosis and neurodevelopmental outcome at 36 months of corrected age and the incidence of tachyarrhythmia after birth, following protocol-defined antenatal therapy for fetal supraventricular tachyarrhythmia.
This was a 3-year follow-up
study of a multicenter
trial that evaluated the efficacy and safety of protocol-defined transplacental treatment for fetal supraventricular tachycardia (SVT) and atrial flutter (AFL). The primary endpoints were mortality and neurodevelopmental impairment (NDI) at 36 months of corrected age. NDI was defined as any of the following outcomes: cerebral palsy, bilateral blindness, bilateral deafness or neurodevelopmental delay. Neurodevelopmental delay was evaluated using appropriate developmental quotient scales, mainly the Kyoto Scale of Psychological Development, or examination by pediatric neurologists. The detection rate of tachyarrhythmia at birth and at 18 and 36 months of corrected age was also evaluated as the secondary endpoint. In addition, the association of NDI at 36 months with perinatal and postnatal factors was analyzed.
Of 50 patients enrolled in the original
trial, one withdrew consent and in two there was fetal death, leaving 47 patients available for enrollment in this follow-up
study. Of these, 45 cases were available for analysis after two infants were lost to follow-up. The mortality rate was 2.2% (1/45) during a median follow-up of 3.2 (range, 2.1-9.4) years. The infant died at the age of 2.1 years. Another infant had missing neurodevelopmental assessment data. In the remaining 43 infants, at 36 months of corrected age, NDI was detected in 9.3% (4/43) overall and in two of three (66.7%) cases with fetal hydrops with subcutaneous edema. Cerebral palsy was noted in two infants with severe subcutaneous edema or ascites at an early gestational age. Neurodevelopmental delay was found in two infants with severe congenital abnormalities (one with tuberous sclerosis and the other with heterotaxy syndrome). Tachyarrhythmia was present in 31.9% (15/47) cases in the neonatal period and decreased to 8.9% (4/45) and 4.5% (2/44) at 18 and 36 months of corrected age, respectively. The median ventricular rate at diagnosis was significantly higher in infants with NDI compared to those without (265 vs 229 bpm; P = 0.003). In infants with NDI, compared to those without, fetal hydrops with subcutaneous edema at diagnosis was more common (50.0% vs 2.6%; P = 0.019) and the duration of fetal effusion was longer (median, 10.5 vs 0 days; P = 0.013). Postnatal arrhythmia and physical development abnormalities were not associated with NDI.
This multicenter 3-year follow-up
study is the first to demonstrate the long-term mortality and morbidity of infants born following protocol-defined transplacental treatment for fetal SVT and AFL. NDI was associated with the presence of fetal hydrops with subcutaneous edema at diagnosis and longer duration of fetal effusion. Neurodevelopmental delay was detected only in infants with severe congenital abnormalities. Therefore, in infants that have undergone antenatal treatment for fetal tachyarrhythmia and in which there are no comorbidities, the risk of NDI is low. However, in those with fetal hydrops with subcutaneous edema and/or associated severe congenital abnormalities, the risk for long-term neurologic morbidity might be considered somewhat increased. © 2022 International Society of Ultrasound in Obstetrics and Gynecology.