Frataxin deficiency and the pathology of Friedreich's Ataxia across tissues

被引:0
作者
Ercanbrack, Wesley S. [1 ]
Ramirez, Mateo [1 ]
Dungan, Austin [1 ]
Gaul, Ella [1 ]
Ercanbrack, Sarah J. [1 ]
Wingert, Rebecca A. [1 ]
机构
[1] Univ Notre Dame, Dept Biol Sci, 100 Galvin Life Sci, Notre Dame, IN 46556 USA
来源
TISSUE BARRIERS | 2025年
关键词
Central nervous system; degeneration; dorsal root; frataxin; Friedreich's Ataxia; heart; kidney; pancreas; peripheral nervous system; scoliosis; NADH-DEHYDROGENASE COMPLEX; NEPHROTIC SYNDROME; DENTATE NUCLEUS; IRON; SCOLIOSIS; MECHANISMS; FEATURES; FAILURE; CELLS; ENTRY;
D O I
10.1080/21688370.2025.2462357
中图分类号
R-3 [医学研究方法]; R3 [基础医学];
学科分类号
1001 ;
摘要
Friedreich's Ataxia (FRDA) is a neurodegenerative disease that affects a variety of different organ systems. The disease is caused by GAA repeat expansions in intron 1 of the Frataxin gene (FXN), which results in a decrease in the expression of the FXN protein. FXN is needed for the biogenesis of iron-sulfur clusters (ISC) which are required by key metabolic processes in the mitochondria. Without ISCs those processes do not occur properly. As a result, reactive oxygen species accumulate, and the mitochondria cease to function. Iron is also thought to accumulate in the cells of certain tissue types. These processes are thought to be intimately related to the pathologies affecting a myriad of tissues in FRDA. Most FRDA patients suffer from loss of motor control, cardiomyopathy, scoliosis, foot deformities, and diabetes. In this review, we discuss the known features of FRDA pathology and the current understanding about the basis of these alterations.
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页数:16
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