ArticleDevelopmental dynamics : an official publication of the American Association of Anatomists2020
Mutational analysis of genes with ureteric progenitor cell-specific expression in branching morphogenesis of the mouse kidney.
Article in Developmental dynamics : an official publication of the American Association of Anatomists, 2020. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 6 papers.
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6 citing papers in PubMed, 9 citations in OpenAlex.
- The role of TMEM59L in colorectal cancer progression and its interaction with the TGF-β/Smad pathway.Frontiers in oncology · 2025Article
- Loss of 3-O-sulfotransferase enzymes, Hs3st3a1 and Hs3st3b1, reduces kidney and glomerular size and disrupts glomerular architecture.Matrix biology : journal of the International Society for Matrix Biology · 2024Article
- The presence of xanthine dehydrogenase is crucial for the maturation of the rat kidneys.Clinical science (London, England : 1979) · 2024Article
- Identification of an energy metabolism-related six-gene signature for distinguishing and forecasting the prognosis of low-grade gliomas.Annals of translational medicine · 2023Article
- Comparative whole-genome transcriptome analysis in renal cell populations reveals high tissue specificity of MAPK/ERK targets in embryonic kidney.BMC biology · 2022Article
- Loss of Hs3st3a1 or Hs3st3b1 enzymes alters heparan sulfate to reduce epithelial morphogenesis and adult salivary gland function.Matrix biology : journal of the International Society for Matrix Biology · 2021Article
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Abstract
backgroundUreteric progenitor cells (UPCs) within the branch tips of the arborizing ureteric epithelium of the kidney's developing collecting system establish the shape and cellular organization of the collecting network, and drive the nephrogenic program through their interactions with nephron progenitor cells. In a previous study, expression screening identified a cohort of genes showing UPC-enriched expression including D17H6S56E-5, Hs3st3a1, Hs3st3b1, and Tmem59l. Each of these is also enriched in branch tips of assembling airways of the developing lungs. Here, we used Crispr-CAS9 directed gene editing to mutate each of these targets to address their potential role(s) in UPC programs.
resultsSingle (D17H6S56E-5 and Tmem59l) and double (Hs3st3a1 and Hs3st3b1) mutants were viable, fertile, and displayed varying frequencies of ureter duplications and no overt lung phenotype. Ureter duplications arise spontaneously through multiple outgrowths of the ureteric bud at the onset of kidney development. Tmem59l mutants and Hs3st3a1/Hs3st3b1 compound mutants showed a weakly penetrant, but statistically significant increase in duplicated ureters compared to C57BL6/J and SW wild-type mouse strains.
conclusionsTmem59l and Hs3st3a1/Hs3st3b1 activities contribute to the regulatory programs restricting ureteric outgrowth in the developing mouse kidney. However, the low penetrance of the observed phenotype precludes a detailed analysis of their specific actions.
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