Section of Pediatric Surgery, Pediatric Liver and Gut Research Group, Children's Hospital, Helsinki University Central Hospital, University of Helsinki, Helsinki, Finland.
Department of Pathology, HUSLAB, Helsinki University Central Hospital, University of Helsinki, Helsinki, Finland.
Ann Surg. 2018 Aug;268(2):332-339. doi: 10.1097/SLA.0000000000002187.
Although liver disease is a major complication of parenteral nutrition (PN) for intestinal failure (IF), its pathogenesis remains unclear. We investigated potential molecular mechanisms of liver injury in pediatric onset IF.
Liver expression of canalicular phospholipid (ABCB4), bile acid (ABCB11), and sterol (ABCG5/8) transporters, their upstream regulators LXR and FXR as well as pro-inflammatory cytokines interleukin-6 (IL6) and tumor necrosis factor (TNF) were investigated among patients with IF [age median 3.8 (IQR 1.2 to 11)] in relation to biochemical and histologic liver injury, PN, serum plant sterols, fibroblast growth factor 19, and α-tocopherol.
Patients receiving PN currently (n = 18) showed more advanced liver injury than patients after weaning off PN (n = 30). Histologic portal inflammation strongly segregated PN-dependent (44%) from weaned off patients (3%, P = 0.001) and coupled with progression of cholestasis and liver fibrosis. Patients with portal inflammation demonstrated markedly induced liver RNA expression of IL6 and TNF, repression of FXR and its canalicular bile transporter target gene RNA expression, including ABCB4 and ABCB11 as well as decreased protein expression of ABCB11 and ABCB4. Furthermore, upregulation of LXR and ABCG5/8 RNA expression was suppressed in patients with portal inflammation. Current PN, increased serum levels of plant sterols stigmasterol, avenasterol, and sitosterol along with serum citrulline, a marker of enterocyte mass, predicted portal inflammation.
In pediatric onset IF, current PN delivery synergistically with intestinal compromise promote liver inflammation, which associates with progression of biochemical and histologic liver injury, while reducing expression of canalicular bile transporters.
虽然肝病是肠衰竭(IF)患者肠外营养(PN)的主要并发症,但发病机制尚不清楚。我们研究了儿科 IF 患者肝损伤的潜在分子机制。
研究了 IF 患者(年龄中位数为 3.8 岁(IQR 1.2 至 11))肝内管腔磷脂(ABCB4)、胆汁酸(ABCB11)和固醇(ABCG5/8)转运体及其上游调节剂 LXR 和 FXR 以及促炎细胞因子白细胞介素 6(IL6)和肿瘤坏死因子(TNF)的表达与肝生化和组织学损伤、PN、血清植物固醇、成纤维细胞生长因子 19 和α-生育酚的关系。
正在接受 PN 治疗的患者(n = 18)的肝损伤比已停用 PN 的患者(n = 30)更严重。组织学门脉炎症强烈地将 PN 依赖性(44%)与断奶患者(3%)分开(P = 0.001),并伴有胆汁淤积和肝纤维化的进展。有门脉炎症的患者表现出明显的肝 RNA 表达的 IL6 和 TNF 增加,FXR 及其管腔胆汁转运体靶基因 RNA 表达的抑制,包括 ABCB4 和 ABCB11 以及 ABCB11 和 ABCB4 的蛋白表达减少。此外,门脉炎症患者的 LXR 和 ABCG5/8 RNA 表达上调受到抑制。目前的 PN、血清甾醇 stigmasterol、avenasterol 和 sitosterol 水平升高以及血清瓜氨酸(一种肠细胞质量的标志物)预测门脉炎症。
在儿科 IF 中,当前 PN 给药与肠道损伤协同作用促进肝脏炎症,这与生化和组织学肝损伤的进展相关,同时降低管腔胆汁转运体的表达。