Ofei Sylvia Y, Texter Karen, Gariepy Cheryl, Smith Sally, Zaidi Ali N, Phelps Christina
University of Kentucky College of Medicine/Kentucky Children's Hospital Department of Pediatrics/Division of Pediatric Gastroenterology, Hepatology and Nutrition 138 Leader Avenue, Rm 206. Lexington, KY 40506. United States of America.
Nationwide Children's Hospital/The Ohio State University Department of Pediatrics/The Heart Center, Division of Pediatric Cardiology 700 Children's Drive. Columbus, OH 43205. United States of America.
Prog Pediatr Cardiol. 2019 Jun;53:21-27. doi: 10.1016/j.ppedcard.2018.09.004. Epub 2018 Oct 6.
Hepatic fibrosis is a significant complication in adult Fontan patients suggesting development as a function of time since the surgery. Children with Fontan circulation are not routinely assessed for development of liver disease. We aimed to evaluate the effectiveness of serologic biomarkers and acoustic radiation force impulse (ARFI) elastography to detect liver disease in pediatric Fontan patients. Patients ≥ 1 year after Fontan operation prospectively had hepatic US with acoustic radiation force impulse and laboratory testing. Clinical cardiac data (echocardiograms, cardiac catheterizations) were reviewed. Statistical analysis was performed using Pearson's correlation coefficient, Wilcoxon rank-sum test and Kruskal-Wallis test. Forty patients were enrolled with median age of 11 years and median time since Fontan of 6.5 years. Platelet count negatively correlated with years since Fontan (p < 0.000). Thrombocytopenia was noted in 15% of patients with the lowest platelet count of 78 K/cu mm, in a patient >10 years from the Fontan (DORV) operation. Alanine transaminase (ALT, p = 0.034) and aspartate aminotransferase (AST, p = 0.009) were higher in patients with Extracardiac Conduit Fontan and not in other Fontan operations. Heterogeneous echotexture on liver ultrasound correlated with years since Fontan (p = 0.022), however all acoustic radiation force impulse values were elevated (> 1.34 m/s) and did not correlate with age, years since Fontan, labs or imaging. FibroSure values did not correlate with years since Fontan. This suggests that ARFI may be elevated due to passive hepatic congestion, limiting its value in this patient population. Additional testing is necessary to identify reliable noninvasive screening modalities for hepatic fibrosis in Fontan patients. Our study is the largest pediatric study to evaluate ARFI in patients after the Fontan operation and showed increased shear wave speed for all patients with no correlation with time since palliation. Decreasing platelet count may indicate the development of liver fibrosis.
肝纤维化是成人Fontan手术患者的一个重要并发症,提示其发生与术后时间有关。Fontan循环的儿童患者通常不常规评估肝脏疾病的发生情况。我们旨在评估血清生物标志物和声辐射力脉冲(ARFI)弹性成像在检测小儿Fontan患者肝脏疾病方面的有效性。Fontan手术后≥1年的患者前瞻性地接受了肝脏超声检查(包括声辐射力脉冲检查)及实验室检测。回顾了临床心脏数据(超声心动图、心导管检查)。使用Pearson相关系数、Wilcoxon秩和检验和Kruskal-Wallis检验进行统计分析。纳入了40例患者,中位年龄为11岁,Fontan手术后的中位时间为6.5年。血小板计数与Fontan手术后的时间呈负相关(p < 0.000)。在15%的患者中发现血小板减少,其中血小板计数最低为78 K/立方毫米,该患者距离Fontan(双心室流出道)手术超过10年。肝外管道Fontan患者的丙氨酸转氨酶(ALT,p = 0.034)和天冬氨酸转氨酶(AST,p = 0.009)较高,而其他Fontan手术患者则不然。肝脏超声的不均匀回声纹理与Fontan手术后的时间相关(p = 0.022),然而所有声辐射力脉冲值均升高(> 1.34 m/s),且与年龄、Fontan手术后的时间、实验室检查或影像学检查均无相关性。FibroSure值与Fontan手术后的时间无相关性。这表明ARFI可能由于被动性肝充血而升高,限制了其在该患者群体中的价值。需要进一步检测以确定用于Fontan患者肝纤维化的可靠无创筛查方法。我们的研究是评估Fontan手术后患者ARFI的最大规模儿科研究,结果显示所有患者的剪切波速度均增加,且与姑息治疗后的时间无关。血小板计数下降可能表明肝纤维化的发生。