过氧化物酶体
生物
肾
内分泌学
内科学
线粒体
肾功能
线粒体生物发生
细胞生物学
生物化学
医学
受体
作者
Camille Ansermet,Gabriel Centeno,Sylvain Pradervand,Dušan Harmáček,Andy Garcia,Jean Daraspe,Sai Kocherlakota,Myriam Baes,Yohan Bignon,Dmitri Firsov
出处
期刊:JCI insight
[American Society for Clinical Investigation]
日期:2022-02-22
卷期号:7 (4)
被引量:7
标识
DOI:10.1172/jci.insight.155836
摘要
Peroxisomes are specialized cellular organelles involved in a variety of metabolic processes. In humans, mutations leading to complete loss of peroxisomes cause multiorgan failure (Zellweger's spectrum disorders, ZSD), including renal impairment. However, the (patho)physiological role of peroxisomes in the kidney remains unknown. We addressed the role of peroxisomes in renal function in mice with conditional ablation of peroxisomal biogenesis in the renal tubule (cKO mice). Functional analyses did not reveal any overt kidney phenotype in cKO mice. However, infant male cKO mice had lower body and kidney weights, and adult male cKO mice exhibited substantial reductions in kidney weight and kidney weight/body weight ratio. Stereological analysis showed an increase in mitochondria density in proximal tubule cells of cKO mice. Integrated transcriptome and metabolome analyses revealed profound reprogramming of a number of metabolic pathways, including metabolism of glutathione and biosynthesis/biotransformation of several major classes of lipids. Although this analysis suggested compensated oxidative stress, challenge with high-fat feeding did not induce significant renal impairments in cKO mice. We demonstrate that renal tubular peroxisomes are dispensable for normal renal function. Our data also suggest that renal impairments in patients with ZSD are of extrarenal origin.
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