Show simple item record

dc.contributor.authorHofherr, A.
dc.contributor.authorSeger, C.
dc.contributor.authorFitzpatrick, F.
dc.date.accessioned2019-06-21T18:46:28Z
dc.date.available2019-06-21T18:46:28Z
dc.date.issued2018
dc.identifier.urihttps://www.scopus.com/inward/record.uri?eid=2-s2.0-85053237333&doi=10.1371%2fjournal.pbio.2005651&partnerID=40&md5=742cba7942a1ea2d69fd0986f9df9d7a
dc.identifier.urihttp://hdl.handle.net/10713/9717
dc.description.abstractCilia 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. Copyright 2018 Hofherr et al. http://creativecommons.org/licenses/by/4.0/.en_US
dc.description.urihttps://dx.doi.org/10.1371/journal.pbio.2005651en_US
dc.language.isoen-USen_US
dc.publisherPublic Library of Scienceen_US
dc.relation.ispartofPLoS Biology
dc.subject.meshCilia--physiologyen_US
dc.subject.meshMitochondria--metabolismen_US
dc.titleThe mitochondrial transporter SLC25A25 links ciliary TRPP2 signaling and cellular metabolismen_US
dc.typeArticleen_US
dc.identifier.doi10.1371/journal.pbio.2005651


This item appears in the following Collection(s)

Show simple item record