Institute of Biochemistry II, Medical Faculty, University of Cologne, Cologne, Germany.
Institute for Human Genetics, University of Cologne, Cologne, Germany.
Proteomics. 2018 Apr;18(7):e1700456. doi: 10.1002/pmic.201700456. Epub 2018 Mar 13.
Autosomal dominant tubulointerstitial kidney disease associated to the MUC1 gene (ADTKD-MUC1; formerly MCKD1) belongs to a heterogeneous group of rare hereditary kidney diseases that is prototypically caused by frameshift mutations in the MUC1 repeat domain. The mutant MUC1 (insC) lacks the transmembrane domaine, exhibits aberant cellular topology, and hence might gain a function during the pathological process. To get insight into potential pathomechanisms we perform differential proteomics of extracellular vesicles shed by renal epithelia into the urine of patients. The study is based on three ADTKD patients and individual controls applying iTRAQ/LC-MS/MS. A total of 796 proteins were identified across all biological and technical replicates, and 298 proteins were quantified. A proportion of 47 proteins were fold-changed species. GO Term Enrichment analysis revealed proteins with significantly changed expression in ADTKD-associated extracellular vesicles as vesicular transport-associated proteins. Among these VTA1 is involved in the endosomal multivesicular body pathway associated with transport to lysosomes or export via exosomes. VTA1 is also claimed to play roles as a cofactor of the AAA ATPases VPS4A and VPS4B in the disassembly of ESCRT III. Protein interaction databases list VPS4B, CHMP2A, and IST1 as VTA1 binding partners. (Data are available via ProteomeXchange with identifier PXD008389.).
常染色体显性遗传性小管间质性肾病与 MUC1 基因(ADTKD-MUC1;前称 MCKD1)相关,属于一组罕见遗传性肾脏疾病的异质性群体,其典型病因是 MUC1 重复结构域的移码突变。突变的 MUC1(insC)缺乏跨膜结构域,表现出异常的细胞拓扑结构,因此可能在病理过程中获得功能。为了深入了解潜在的发病机制,我们对肾脏上皮细胞分泌到尿液中的细胞外囊泡进行了差异蛋白质组学研究。该研究基于 3 名 ADTKD 患者和个体对照,采用 iTRAQ/LC-MS/MS 进行分析。在所有生物学和技术重复中总共鉴定出 796 种蛋白质,其中 298 种蛋白质被定量。有 47 种蛋白质的丰度发生了变化。GO 术语富集分析显示,ADTKD 相关细胞外囊泡中表达发生显著变化的蛋白质与囊泡运输相关蛋白有关。其中,VTA1 参与与溶酶体或通过外泌体输出相关的内体多泡体途径。VTA1 还被认为在 ESCRT III 的解组装中作为 AAA ATPases VPS4A 和 VPS4B 的辅助因子发挥作用。蛋白质相互作用数据库列出了 VPS4B、CHMP2A 和 IST1 作为 VTA1 的结合伙伴。(数据可通过 ProteomeXchange 以标识符 PXD008389. 获取。)