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- Title
The mitochondrial transporter SLC25A25 links ciliary TRPP2 signaling and cellular metabolism.
- Authors
Hofherr, Alexis; Seger, Claudia; Fitzpatrick, Fiona; Busch, Tilman; Michel, Elisabeth; Luan, Jingting; Osterried, Lea; Linden, Frieder; Kramer-Zucker, Albrecht; Wakimoto, Barbara; Schütze, Conny; Wiedemann, Nils; Artati, Anna; Adamski, Jerzy; Walz, Gerd; Kunji, Edmund R. S.; Montell, Craig; Watnick, Terry; Köttgen, Michael
- 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.
- Subjects
CELLULAR signal transduction; MITOCHONDRIA; CELL metabolism; TRP channels; POLYCYSTINS
- Publication
PLoS Biology, 2018, Vol 16, Issue 8, p1
- ISSN
1544-9173
- Publication type
Article
- DOI
10.1371/journal.pbio.2005651