Murin Peter, Sinzobahamvya Nicodème, Blaschczok Hedwig Ch, Photiadis Joachim, Haun Christoph, Asfour Boulos, Hraska Viktor
Deutsches Kinderherzzentrum Sankt Augustin, Sankt Augustin, Germany.
Thorac Cardiovasc Surg. 2012 Apr;60(3):215-20. doi: 10.1055/s-0031-1298061. Epub 2012 Jan 17.
Patients with combined aortopulmonary window (APW) and interrupted aortic arch (IAA) malformations are rarely seen. We reviewed cases with such association with emphasis on surgical management and long-term outcome.
109 patients with IAA and 25 patients with APW were operated upon between 1981 and 2011. The clinical records, operation and follow-up data were analyzed. Long-term outcome was completed with the help of either outpatient data or inquiring. Related literature was investigated.
Combined APW/IAA was found in 8 cases: 7.3% (8/109) of those with IAA diagnosis and 32% (8/25) with APW. All APWs were proximal. 7 patients had interruption type A. A ventricular septal defect (VSD) was associated in one case only. Median age at surgery was 10 days. The two first patients were operated upon without cardio-pulmonary bypass (CPB) and one died during operation. The other six underwent single stage approach under CPB with no death: overall early mortality of 12.5% (⅛). APW was closed with one or two patches; aortic arch was reconstructed either directly (extended end-to-side anastomosis) or by patch augmentation. There was no late death and no reoperation during mean follow-up of 118 months (range 1-360 months). Six patients were in functional NYHA class I, the remainder in class II. The cumulative APW/IAA incidence from literature varies between 4.9% (56/1105) and 22.2% (42/189). Early mortality reaches 15.1% (8/53) (95% CI: 6.8% - 27.6%).
Neonatal repair with thorough mobilization of the aortic arch and extended end-to-side anastomosis or use of patch augmentation carries potential for the best early and late outcome for combined APW/IAA malformation.
合并主肺动脉窗(APW)和主动脉弓中断(IAA)畸形的患者极为罕见。我们回顾了此类关联病例,重点关注手术治疗及长期预后。
1981年至2011年间,对109例IAA患者和25例APW患者进行了手术。分析临床记录、手术及随访数据。通过门诊数据或询问完成长期预后评估。检索相关文献。
发现8例合并APW/IAA:IAA诊断患者中的7.3%(8/109)以及APW患者中的32%(8/25)。所有APW均为近端型。7例患者为A型中断。仅1例合并室间隔缺损(VSD)。手术时的中位年龄为10天。最初的2例患者在无体外循环(CPB)下进行手术,1例在术中死亡。其他6例在CPB下接受一期手术,无死亡:总体早期死亡率为12.5%(1/8)。APW用一片或两片补片关闭;主动脉弓直接重建(延长端到侧吻合)或用补片扩大。在平均118个月(范围1 - 360个月)的随访期间,无晚期死亡及再次手术情况。6例患者心功能为纽约心脏协会(NYHA)I级,其余为II级。文献报道的APW/IAA累积发病率在4.9%(56/1105)至22.2%(42/189)之间。早期死亡率达15.1%(8/53)(95%可信区间:6.8% - 27.6%)。
对于合并APW/IAA畸形,新生儿期进行彻底游离主动脉弓并采用延长端到侧吻合术或补片扩大术进行修复,有望获得最佳的早期和晚期预后。