de Lange Charlotte, Möller Thomas, Hebelka Hanna
Department of Pediatric Radiology, Queen Silvia Children's Hospital, Sahlgrenska University Hospital, Gothenburg, Sweden.
Institution of Clinical Sciences, Sahlgrenska Academy, University of Gothenburg, Gothenburg, Sweden.
Front Pediatr. 2023 Mar 3;11:1100514. doi: 10.3389/fped.2023.1100514. eCollection 2023.
The Fontan operation is a lifesaving procedure for patients with functional single-ventricle congenital heart disease, where hypoplastic left heart syndrome is the most frequent anomaly. Hemodynamic changes following Fontan circulation creation are now increasingly recognized to cause multiorgan affection, where the development of a chronic liver disease, Fontan-associated liver disease (FALD), is one of the most important morbidities. Virtually, all patients with a Fontan circulation develop liver congestion, resulting in fibrosis and cirrhosis, and most patients experience childhood onset. FALD is a distinctive type of congestive hepatopathy, and its pathogenesis is thought to be a multifactorial process driven by increased nonpulsatile central venous pressure and decreased cardiac output, both of which are inherent in the Fontan circulation. In the advanced stage of liver injury, complications of portal hypertension often occur, and there is a risk of developing secondary liver cancer, reported at young age. However, FALD develops with few clinical symptoms, a surprisingly variable degree of severity in liver disease, and with little relation to poor cardiac function. The disease mechanisms and modifying factors of its development are still not fully understood. As one of the more important noncardiac complications of the Fontan circulation, FALD needs to be diagnosed in a timely manner with a structured monitoring scheme of disease development, early detection of malignancy, and determination of the optimal time point for transplantation. There is also a clear need for consensus on the best surveillance strategy for FALD. In this regard, imaging plays an important role together with clinical scoring systems, biochemical workups, and histology. Patients operated on with a Fontan circulation are generally followed up in cardiology units. Ultimately, the resulting multiorgan affection requires a multidisciplinary team of healthcare personnel to address the different organ complications. This article discusses the current concepts, diagnosis, and management of FALD, with special emphasis on the role of different imaging techniques in the diagnosis and monitoring of disease progression, as well as current recommendations for liver disease surveillance.
Fontan手术是功能性单心室先天性心脏病患者的一种挽救生命的手术,其中左心发育不全综合征是最常见的异常情况。现在越来越认识到,Fontan循环建立后的血流动力学变化会导致多器官受累,其中慢性肝病即Fontan相关肝病(FALD)的发展是最重要的发病情况之一。实际上,所有接受Fontan循环手术的患者都会出现肝充血,进而导致纤维化和肝硬化,且大多数患者在儿童期发病。FALD是一种独特类型的充血性肝病,其发病机制被认为是一个多因素过程,由非搏动性中心静脉压升高和心输出量降低驱动,而这两者都是Fontan循环所固有的。在肝损伤的晚期,常发生门静脉高压并发症,且有在年轻时发生继发性肝癌的风险。然而,FALD的发展临床症状较少,肝病严重程度差异惊人,且与心功能不良关系不大。其发病的疾病机制和调节因素仍未完全了解。作为Fontan循环更重要的非心脏并发症之一,FALD需要通过疾病发展的结构化监测方案、恶性肿瘤的早期检测以及移植最佳时间点的确定来及时诊断。对于FALD的最佳监测策略也显然需要达成共识。在这方面,影像学与临床评分系统、生化检查和组织学一起发挥着重要作用。接受Fontan循环手术的患者通常在心脏病科进行随访。最终,由此产生的多器官受累需要多学科医疗团队来处理不同器官的并发症。本文讨论了FALD的当前概念、诊断和管理,特别强调了不同成像技术在疾病诊断和进展监测中的作用,以及当前肝病监测的建议。