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The DNA damage protein and transcription factor Atmin (Asciz) is required for both lung tubulogenesis and ciliogenesis. Like the lungs, kidneys contain a tubular network that is critical for their function and in addition, renal ciliary dysfunction has been implicated in the pathogenesis of cystic kidney disease. Using the  Atmin  mouse mutant  Gasping6 (Gpg6 ), we investigated kidney development and found it severely disrupted with reduced branching morphogenesis, resulting in fewer epithelial structures being formed. Unexpectedly, transcriptional levels of key cilia associated genes were not altered in Atmin  Gpg6/Gpg6   kidneys. Instead,  Gpg6  homozygous kidneys exhibited altered cytoskeletal organization and modulation of Wnt signaling pathway molecules, including β-catenin and non-canonical Wnt/planar cell polarity (PCP) pathway factors, such as  Daam2  and Vangl2. Wnt signaling is important for kidney development and perturbation of Wnt signaling pathways can result in cystic, and other, renal abnormalities. In common with other PCP pathway mutants,  Atmin Gpg6/Gpg6   mice displayed a shortened rostral-caudal axis and mis-oriented cell division. Moreover, intercrosses between  Atmin Gpg6/+   and  Vangl2 Lp/+   mice revealed a genetic interaction between  Atmin  and  Vangl2 . Thus we show for the first time that  Atmin  is critical for normal kidney development and we present evidence that mechanistically,  Atmin  modifies Wnt signaling pathways, specifically placing it as a novel effector molecule in the non-canonical Wnt/PCP pathway. The identification of a novel modulator of Wnt signaling has important implications for understanding the pathobiology of renal disease.

Atmin mediates kidney morphogenesis by modulating Wnt signaling