Cargando…
The chloride antiporter CLCN7 is a modifier of lysosome dysfunction in FIG4 and VAC14 mutants
The phosphatase FIG4 and the scaffold protein VAC14 function in the biosynthesis of PI(3,5)P(2), a signaling lipid that inhibits the lysosomal chloride transporter ClC-7. Loss-of-function mutations of FIG4 and VAC14 reduce PI(3,5)P(2) and result in lysosomal disorders characterized by accumulation o...
Autores principales: | Cao, Xu, Lenk, Guy M., Mikusevic, Vedrana, Mindell, Joseph A., Meisler, Miriam H. |
---|---|
Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
Public Library of Science
2023
|
Materias: | |
Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10328317/ https://www.ncbi.nlm.nih.gov/pubmed/37363915 http://dx.doi.org/10.1371/journal.pgen.1010800 |
Ejemplares similares
-
Tonic inhibition of the chloride/proton antiporter ClC-7 by PI(3,5)P2 is crucial for lysosomal pH maintenance
por: Leray, Xavier, et al.
Publicado: (2022) -
Altered phenotypes due to genetic interaction between the mouse phosphoinositide biosynthesis genes Fig4 and Pip4k2c
por: Cao, Xu, et al.
Publicado: (2023) -
Murine Fig4 is dispensable for muscle development but required for muscle function
por: Reifler, Aaron, et al.
Publicado: (2013) -
The Vac14p–Fig4p complex acts independently of Vac7p and couples PI3,5P(2) synthesis and turnover
por: Duex, Jason E., et al.
Publicado: (2006) -
Neuropathology of childhood‐onset basal ganglia degeneration caused by mutation of VAC14
por: Stutterd, Chloe, et al.
Publicado: (2017)