Escobar-Diaz Maria C, Tworetzky Wayne, Friedman Kevin, Lafranchi Terra, Fynn-Thompson Francis, Alexander Mark E, Mah Douglas Y
Department of Cardiology, Boston Children's Hospital, 300 Longwood Avenue, Boston, MA, 02115, USA,
Pediatr Cardiol. 2014 Aug;35(6):906-13. doi: 10.1007/s00246-014-0874-x. Epub 2014 Feb 9.
Congenital atrioventricular (AV) block is commonly associated with heterotaxy syndrome; together they have reportedly low survival rates (10-25%). However, information about perinatal outcome and predictors of non-survival after prenatal diagnosis of this association is scarce. Therefore, we studied fetuses with heterotaxy syndrome and bradycardia or AV-block diagnosed between 1995 and 2011, and analyzed pre and post-natal variables. The primary outcome was death and the secondary outcome was pacemaker placement. Of the 154 fetuses with heterotaxy syndrome, 91 had polysplenia syndrome, 22/91(24%) with bradycardia or AV-block. Thirteen (59%) patients had sinus bradycardia at diagnosis, 8 (36%) complete AV block, and 1 (5%) second-degree AV-block. Three patients elected for termination of pregnancy (3/22, 14%), 4 had spontaneous fetal demise (4/22, 18%), and 15 (15/22, 68%) were live-born. Of the fetuses with bradycardia/AV-block, 30% presented with hydrops, 20% had ventricular rates <55 beats/min, and 10% had cardiac dysfunction. Excluding termination of pregnancy, 15/19 fetuses (79%) survived to birth. Among the 15 live-born patients, 4 had bradycardia and 11 had AV-block. A further 3 patients died in infancy, all with AV-block who required pacemakers in the neonatal period. Thus, the 1-year survival rate, excluding termination of pregnancy, was 63% (12/19). Of the remaining 12 patients, 9 required pacemaker. Predictors of perinatal death included hydrops (p < 0.0001), ventricular dysfunction (p = 0.002), prematurity (p = 0.04), and low ventricular rates (p = 0.04). In conclusion, we found a higher survival rate (63%) than previously published in patients with heterotaxy syndrome and AV block or bradycardia diagnosed prenatally. Hydrops, cardiac dysfunction, prematurity and low ventricular rates were predictors of death.
先天性房室传导阻滞通常与内脏异位综合征相关;据报道,二者共同存在时生存率较低(10%-25%)。然而,关于产前诊断出这种关联后的围产期结局及非存活预测因素的信息却很匮乏。因此,我们研究了1995年至2011年间诊断为内脏异位综合征且伴有心动过缓或房室传导阻滞的胎儿,并分析了产前和产后变量。主要结局是死亡,次要结局是起搏器植入。在154例内脏异位综合征胎儿中,91例患有多脾综合征,其中22/91(24%)伴有心动过缓或房室传导阻滞。13例(59%)患者诊断时为窦性心动过缓,8例(36%)为完全性房室传导阻滞,1例(5%)为二度房室传导阻滞。3例患者选择终止妊娠(3/22,14%),4例胎儿自然死亡(4/22,18%),15例(15/22,68%)存活出生。在伴有心动过缓/房室传导阻滞的胎儿中,30%出现水肿,20%心室率<55次/分钟,10%存在心脏功能障碍。排除终止妊娠的情况,15/19例胎儿(79%)存活至出生。在15例存活出生的患者中,4例有心动过缓,11例有房室传导阻滞。另有3例患者在婴儿期死亡,均为房室传导阻滞,且在新生儿期需要植入起搏器。因此,排除终止妊娠的情况,1年生存率为63%(12/19)。在其余12例患者中,9例需要植入起搏器。围产期死亡的预测因素包括水肿(p<0.0001)、心室功能障碍(p = 0.002)、早产(p = 0.04)和心室率低(p = 0.04)。总之,我们发现产前诊断为内脏异位综合征且伴有房室传导阻滞或心动过缓的患者生存率(63%)高于先前报道。水肿、心脏功能障碍、早产和心室率低是死亡的预测因素。