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法布里病肾移植的长期结局。

Long-term Outcomes of Kidney Transplantation in Fabry Disease.

机构信息

Department of Cardiology, Inselspital, Bern University Hospital, Bern, Switzerland.

Department of Internal Medicine, University Hospital Zurich, Zurich, Switzerland.

出版信息

Transplantation. 2018 Nov;102(11):1924-1933. doi: 10.1097/TP.0000000000002252.

Abstract

BACKGROUND

Fabry disease (FD) is a rare X-linked lysosomal storage disorder caused by mutations in the α-galactosidase A gene that obliterate or markedly reduce α-galactosidase A activity. This results in the systemic accumulation of its glycosphingolipid substrates in body fluids and organs, including the kidney. Fabry nephropathy can lead to end-stage renal disease requiring kidney transplantation. Little is known about its long-term outcomes and the overall patient survival after kidney transplantation.

METHODS

Here, we report 17 Fabry patients (15 male and 2 female subjects) who received kidney transplants and their long-term treatment and follow-up at 4 specialized Fabry centers.

RESULTS

The posttransplant follow-up ranged to 25 years, with a median of 11.5 (range, 0.8-25.5] years. Graft survival was similar, and death-censored graft survival was superior to matched controls. Fabry patients died with functioning kidneys, mostly from cardiac causes. In 2 male subjects 14 and 23 years posttransplant, the grafts had a few typical FD lamellar inclusions, presumably originating from invading host macrophages and vascular endothelial cells.

CONCLUSIONS

We conclude that kidney transplantation has an excellent long-term outcome in FD.

摘要

背景

法布瑞病(FD)是一种罕见的 X 连锁溶酶体贮积症,由α-半乳糖苷酶 A 基因突变引起,导致α-半乳糖苷酶 A 活性完全或显著降低。这会导致其糖脂底物在体液和器官中全身积累,包括肾脏。法布瑞肾病可导致终末期肾病,需要进行肾移植。关于其长期结局以及肾移植后患者的总体存活率知之甚少。

方法

在这里,我们报告了 17 名接受肾移植的法布瑞病患者(15 名男性和 2 名女性)及其在 4 个专门的法布瑞病中心的长期治疗和随访情况。

结果

移植后的随访时间最长可达 25 年,中位数为 11.5 年(范围,0.8-25.5 年)。移植物存活率相似,且经死亡校正的移植物存活率优于匹配的对照组。法布瑞病患者在有功能的肾脏中死亡,主要死于心脏原因。在移植后 14 年和 23 年的 2 名男性患者中,移植物中存在一些典型的 FD 层状包涵体,推测源自入侵的宿主巨噬细胞和血管内皮细胞。

结论

我们得出结论,肾移植在 FD 中具有极好的长期结局。

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