Unseld Bettina, Stiller Brigitte, Borth-Bruhns Thomas, du Bois Florian, Kroll Johannes, Grohmann Jochen, Fleck Thilo
Department of Congenital Heart Disease and Pediatric Cardiology, University Heart Centre Freiburg, Mathildenstrasse 1, 79106, Freiburg, Germany.
Nachsorgeklinik Tannheim, Rehabilitationsklinik, VS-Tannheim, Germany.
Pediatr Cardiol. 2017 Aug;38(6):1155-1161. doi: 10.1007/s00246-017-1632-7. Epub 2017 May 22.
Protein-losing enteropathy (PLE) and plastic bronchitis (PB) are major causes of long-term mortality after Fontan operation. The objective of this study was to determine early clinical risk factors before the onset of PLE and PB. In a cohort study, 106 Fontan patients between 2005 and 2013 were examined. A median of 5.3 (1.5-8.5) years later, follow-up questionnaires were used to group the patients in a PLE or PB group (n = 14) and a non-PLE/PB group (n = 92). Prevalence of PLE was 9.4% (n = 10) and of PB 3.8% (n = 4). At follow-up, five patients (4.7%) died of PLE or PB. Median age at death was 6.2 years (IQR 10.5, 95% CI 5.3-23.4). We observed no significant group differences in gender distribution (p = 0.73), ventricular morphology (p = 0.87), surgical technique (p = 0.64), conduit fenestration (p = 0.34), age at Fontan operation (p = 0.54), and need for diuretics (p = 0.56). Hypoplastic left heart syndrome was more frequent in the PLE/PB group 50 vs. 22.8% (p = 0.03) OR 3.4 (95% CI 1.1-10.8). The modified Glenn procedure was performed at a median age of 4 months (IQR 4.0) in the PLE/PB group versus 8 months (IQR 8.0) in the non-PLE/PB group (p = 0.01). The early Glenn procedure and hypoplastic left heart syndrome may be associated with the development of PLE and PB.
蛋白丢失性肠病(PLE)和塑形支气管炎(PB)是Fontan手术术后长期死亡的主要原因。本研究的目的是确定PLE和PB发病前的早期临床风险因素。在一项队列研究中,对2005年至2013年间的106例Fontan患者进行了检查。在中位时间5.3(1.5 - 8.5)年后,使用随访问卷将患者分为PLE或PB组(n = 14)和非PLE/PB组(n = 92)。PLE的患病率为9.4%(n = 10),PB的患病率为3.8%(n = 4)。在随访时,5例患者(4.7%)死于PLE或PB。死亡时的中位年龄为6.2岁(四分位间距10.5,95%置信区间5.3 - 23.4)。我们观察到在性别分布(p = 0.73)、心室形态(p = 0.87)、手术技术(p = 0.64)、管道开窗(p = 0.34)、Fontan手术时的年龄(p = 0.54)以及利尿剂使用需求(p = 0.56)方面,两组之间无显著差异。左心发育不全综合征在PLE/PB组中更为常见,为50%,而非PLE/PB组为22.8%(p = 0.03),比值比为3.4(95%置信区间1.1 - 10.8)。PLE/PB组进行改良Glenn手术的中位年龄为4个月(四分位间距4.0),而非PLE/PB组为8个月(四分位间距8.0)(p = 0.01)。早期Glenn手术和左心发育不全综合征可能与PLE和PB的发生有关。