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PLoS Biol ; 16(8): e2005651, 2018 08.
Article in English | MEDLINE | ID: mdl-30080851

ABSTRACT

Cilia are organelles specialized in movement and signal transduction. The ciliary transient receptor potential ion channel polycystin-2 (TRPP2) controls elementary cilia-mediated physiological functions ranging from male fertility and kidney development to left-right patterning. However, the molecular components translating TRPP2 channel-mediated Ca2+ signals into respective physiological functions are unknown. Here, we show that the Ca2+-regulated mitochondrial ATP-Mg/Pi solute carrier 25 A 25 (SLC25A25) acts downstream of TRPP2 in an evolutionarily conserved metabolic signaling pathway. We identify SLC25A25 as an essential component in this cilia-dependent pathway using a genome-wide forward genetic screen in Drosophila melanogaster, followed by a targeted analysis of SLC25A25 function in zebrafish left-right patterning. Our data suggest that TRPP2 ion channels regulate mitochondrial SLC25A25 transporters via Ca2+ establishing an evolutionarily conserved molecular link between ciliary signaling and mitochondrial metabolism.


Subject(s)
Amino Acid Transport Systems, Acidic/metabolism , Calcium-Binding Proteins/metabolism , Cilia/metabolism , TRPP Cation Channels/metabolism , Animals , Antiporters/metabolism , Calcium/metabolism , Calcium Channels/metabolism , Drosophila melanogaster/metabolism , Heterozygote , Humans , Ion Channels/metabolism , Mitochondrial Membrane Transport Proteins/metabolism , Mitochondrial Proteins/metabolism , Signal Transduction , Zebrafish
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