Department of Pediatrics University of Alberta Edmonton Alberta Canada.
Glenrose Rehabilitation Hospital Edmonton Alberta Canada.
J Am Heart Assoc. 2020 Feb 18;9(4):e013632. doi: 10.1161/JAHA.119.013632. Epub 2020 Feb 11.
Background Management of patients with hypoplastic left heart syndrome has benefited from advancements in medical and surgical care. Outcomes have improved, although survival and long-term functional and cognitive deficits remain a concern. Methods and Results This is a cohort study of all consecutive patients with hypoplastic left heart syndrome undergoing surgical palliation at a single center. We aimed to examine demographic and perioperative factors from each surgical stage for their association with survival and neurocognitive outcomes. A total of 117 consecutive patients from 1996 to 2010 underwent surgical palliation. Seventy patients (60%) survived to the Fontan stage and 68 patients (58%) survived to undergo neurocognitive assessment at a mean (SD) age of 56.6 months (6.4 months). Full-scale, performance, and verbal intelligence quotient, as well as visual-motor integration mean (SD) scores were 86.7 (16.1), 86.3 (15.8), 88.8 (17.2), and 83.2 (14.8), respectively. On multivariable analysis, older age at Fontan, sepsis peri-Norwood, lowest arterial partial pressure of oxygen postbidirectional cavopulmonary anastomosis, and presence of neuromotor disability pre-Fontan were strongly associated with lower scores for all intelligence quotient domains. Older age at Fontan and sepsis peri-Norwood remained associated with lower scores for all intelligence quotient domains in a subgroup analysis excluding patients with disability pre-Fontan or with chromosomal abnormalities. Conclusions Older age at Fontan and sepsis are among independent predictors of poor neurocognitive outcomes for patients with hypoplastic left heart syndrome. Further studies are required to identify the appropriate age range for Fontan completion, balancing a lower risk of acute and long-term hemodynamic complications while optimizing long-term neurocognitive outcomes.
左心发育不全综合征患者的管理得益于医学和外科治疗的进步。尽管生存和长期的功能和认知缺陷仍然是一个问题,但结果已经得到改善。
这是一项在单中心对所有接受左心发育不全综合征手术姑息治疗的连续患者进行的队列研究。我们旨在检查每个手术阶段的人口统计学和围手术期因素,以了解其与生存率和神经认知结果的关系。1996 年至 2010 年间,共有 117 例连续患者接受了手术姑息治疗。70 例(60%)患者存活至 Fontan 阶段,68 例(58%)患者存活至接受神经认知评估,平均(标准差)年龄为 56.6 个月(6.4 个月)。全量表、表现量表和言语智商以及视觉运动整合的平均(标准差)得分分别为 86.7(16.1)、86.3(15.8)、88.8(17.2)和 83.2(14.8)。多变量分析显示,Fontan 时年龄较大、Norwood 围手术期败血症、双向腔肺吻合术后最低动脉部分氧分压和 Fontan 前存在运动神经功能障碍与所有智商领域的得分较低密切相关。在排除 Fontan 前存在残疾或染色体异常的患者的亚组分析中,Fontan 时年龄较大和 Norwood 围手术期败血症仍然与所有智商领域的得分较低相关。
Fontan 时年龄较大和败血症是左心发育不全综合征患者神经认知结局不良的独立预测因素。需要进一步研究确定 Fontan 完成的适当年龄范围,在降低急性和长期血液动力学并发症风险的同时,优化长期神经认知结局。