The mitochondrial transporter SLC25A25 links ciliary TRPP2 signaling and cellular metabolism
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
- Location
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Deutsche Nationalbibliothek Frankfurt am Main
- Extent
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Online-Ressource
- Language
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Englisch
- Notes
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PLOS Biology. - 16, 8 (2018) , e2005651, ISSN: 1545-7885
- Event
-
Veröffentlichung
- (where)
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Freiburg
- (who)
-
Universität
- (when)
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2019
- Creator
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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, Edmundus Richardus Stephanus
Montell, Craig
Watnick, Terry
Köttgen, Michael
- Contributor
- DOI
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10.1371/journal.pbio.2005651
- URN
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urn:nbn:de:bsz:25-freidok-1487371
- Rights
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Kein Open Access; Der Zugriff auf das Objekt ist unbeschränkt möglich.
- Last update
-
15.08.2025, 7:37 AM CEST
Data provider
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Associated
- 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, Edmundus Richardus Stephanus
- Montell, Craig
- Watnick, Terry
- Köttgen, Michael
- Klinik für Innere Medizin IV. Freiburg im Breisgau
- Universität
Time of origin
- 2019
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