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000164661 037__ $$aDZNE-2022-01191
000164661 041__ $$aEnglish
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000164661 1001_ $$aHansen, Jan N$$b0
000164661 245__ $$aA cAMP signalosome in primary cilia drives gene expression and kidney cyst formation
000164661 260__ $$aHoboken, NJ [u.a.]$$bWiley$$c2022
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000164661 520__ $$aThe primary cilium constitutes an organelle that orchestrates signal transduction independently from the cell body. Dysregulation of this intricate molecular architecture leads to severe human diseases, commonly referred to as ciliopathies. However, the molecular underpinnings how ciliary signaling orchestrates a specific cellular output remain elusive. By combining spatially resolved optogenetics with RNA sequencing and imaging, we reveal a novel cAMP signalosome that is functionally distinct from the cytoplasm. We identify the genes and pathways targeted by the ciliary cAMP signalosome and shed light on the underlying mechanisms and downstream signaling. We reveal that chronic stimulation of the ciliary cAMP signalosome transforms kidney epithelia from tubules into cysts. Counteracting this chronic cAMP elevation in the cilium by small molecules targeting activation of phosphodiesterase-4 long isoforms inhibits cyst growth. Thereby, we identify a novel concept of how the primary cilium controls cellular functions and maintains tissue integrity in a specific and spatially distinct manner and reveal novel molecular components that might be involved in the development of one of the most common genetic diseases, polycystic kidney disease.
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000164661 650_7 $$2Other$$aCREB
000164661 650_7 $$2Other$$aPKD
000164661 650_7 $$2Other$$acAMP
000164661 650_7 $$2Other$$aoptogenetics
000164661 650_7 $$2Other$$aprimary cilia
000164661 650_2 $$2MeSH$$aCilia: metabolism
000164661 650_2 $$2MeSH$$aCysts: metabolism
000164661 650_2 $$2MeSH$$aGene Expression
000164661 650_2 $$2MeSH$$aHumans
000164661 650_2 $$2MeSH$$aKidney
000164661 650_2 $$2MeSH$$aPolycystic Kidney Diseases: genetics
000164661 650_2 $$2MeSH$$aPolycystic Kidney Diseases: metabolism
000164661 7001_ $$aKaiser, Fabian$$b1
000164661 7001_ $$aLeyendecker, Philipp$$b2
000164661 7001_ $$aStüven, Birthe$$b3
000164661 7001_ $$aKrause, Jens-Henning$$b4
000164661 7001_ $$aDerakhshandeh, Fatemeh$$b5
000164661 7001_ $$aIrfan, Jaazba$$b6
000164661 7001_ $$00000-0002-9016-5989$$aSroka, Tommy J$$b7
000164661 7001_ $$aPreval, Kenley M$$b8
000164661 7001_ $$aDesai, Paurav B$$b9
000164661 7001_ $$0P:(DE-2719)9000840$$aKraut, Michael$$b10$$udzne
000164661 7001_ $$0P:(DE-2719)2812165$$aTheis, Heidi$$b11$$udzne
000164661 7001_ $$0P:(DE-2719)2812302$$aDrews, Anna-Dorothee$$b12$$udzne
000164661 7001_ $$0P:(DE-2719)9000846$$ade Domenico, Elena$$b13$$udzne
000164661 7001_ $$0P:(DE-2719)2812735$$aHändler, Kristian$$b14$$udzne
000164661 7001_ $$00000-0002-6285-8796$$aPazour, Gregory J$$b15
000164661 7001_ $$aHenderson, David J P$$b16
000164661 7001_ $$00000-0003-1427-9412$$aMick, David U$$b17
000164661 7001_ $$00000-0003-4800-6332$$aWachten, Dagmar$$b18
000164661 773__ $$0PERI:(DE-600)2025376-X$$a10.15252/embr.202154315$$gVol. 23, no. 8$$n8$$pe54315$$tEMBO reports$$v23$$x1469-221X$$y2022
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