PAX2 is dispensable for in vitro nephron formation from human induced pluripotent stem cells

Abstract The kidney is formed by reciprocal interactions between the nephron progenitor and the ureteric bud, the former of which gives rise to the epithelia of nephrons consisting of glomeruli and renal tubules. The transcription factor PAX2 is essential for this mesenchymal-to-epithelial transitio...

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Autores principales:	Yusuke Kaku, Atsuhiro Taguchi, Shunsuke Tanigawa, Fahim Haque, Tetsushi Sakuma, Takashi Yamamoto, Ryuichi Nishinakamura
Formato:	article
Lenguaje:	EN
Publicado:	Nature Portfolio 2017
Materias:	Medicine R Science Q
Acceso en línea:	https://doaj.org/article/5c93a9331aaf4abe88ba9f48b1f8daa1
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spelling	oai:doaj.org-article:5c93a9331aaf4abe88ba9f48b1f8daa12021-12-02T15:06:14ZPAX2 is dispensable for in vitro nephron formation from human induced pluripotent stem cells10.1038/s41598-017-04813-32045-2322https://doaj.org/article/5c93a9331aaf4abe88ba9f48b1f8daa12017-07-01T00:00:00Zhttps://doi.org/10.1038/s41598-017-04813-3https://doaj.org/toc/2045-2322Abstract The kidney is formed by reciprocal interactions between the nephron progenitor and the ureteric bud, the former of which gives rise to the epithelia of nephrons consisting of glomeruli and renal tubules. The transcription factor PAX2 is essential for this mesenchymal-to-epithelial transition of nephron progenitors, as well as ureteric bud lineage development, in mice. PAX2 mutations in humans cause renal coloboma syndrome. We previously reported the induction of nephron progenitors and three-dimensional nephron structures from human induced pluripotent stem (iPS) cells. Here we generate iPS cells lacking PAX2, and address the role of PAX2 in our in vitro induction protocol. While PAX2-null human nephron progenitors were properly formed, they unexpectedly became epithelialised to form glomeruli and renal tubules. However, the mutant glomerular parietal epithelial cells failed to transit to the squamous morphology, retaining the shape and markers of columnar epithelia. Therefore, PAX2 is dispensable for mesenchymal-to-epithelial transition of nephron progenitors, but is required for morphological development of glomerular parietal epithelial cells, during nephron formation from human iPS cells in vitro.Yusuke KakuAtsuhiro TaguchiShunsuke TanigawaFahim HaqueTetsushi SakumaTakashi YamamotoRyuichi NishinakamuraNature PortfolioarticleMedicineRScienceQENScientific Reports, Vol 7, Iss 1, Pp 1-12 (2017)
institution	DOAJ
collection	DOAJ
language	EN
topic	Medicine R Science Q
spellingShingle	Medicine R Science Q Yusuke Kaku Atsuhiro Taguchi Shunsuke Tanigawa Fahim Haque Tetsushi Sakuma Takashi Yamamoto Ryuichi Nishinakamura PAX2 is dispensable for in vitro nephron formation from human induced pluripotent stem cells
description	Abstract The kidney is formed by reciprocal interactions between the nephron progenitor and the ureteric bud, the former of which gives rise to the epithelia of nephrons consisting of glomeruli and renal tubules. The transcription factor PAX2 is essential for this mesenchymal-to-epithelial transition of nephron progenitors, as well as ureteric bud lineage development, in mice. PAX2 mutations in humans cause renal coloboma syndrome. We previously reported the induction of nephron progenitors and three-dimensional nephron structures from human induced pluripotent stem (iPS) cells. Here we generate iPS cells lacking PAX2, and address the role of PAX2 in our in vitro induction protocol. While PAX2-null human nephron progenitors were properly formed, they unexpectedly became epithelialised to form glomeruli and renal tubules. However, the mutant glomerular parietal epithelial cells failed to transit to the squamous morphology, retaining the shape and markers of columnar epithelia. Therefore, PAX2 is dispensable for mesenchymal-to-epithelial transition of nephron progenitors, but is required for morphological development of glomerular parietal epithelial cells, during nephron formation from human iPS cells in vitro.
format	article
author	Yusuke Kaku Atsuhiro Taguchi Shunsuke Tanigawa Fahim Haque Tetsushi Sakuma Takashi Yamamoto Ryuichi Nishinakamura
author_facet	Yusuke Kaku Atsuhiro Taguchi Shunsuke Tanigawa Fahim Haque Tetsushi Sakuma Takashi Yamamoto Ryuichi Nishinakamura
author_sort	Yusuke Kaku
title	PAX2 is dispensable for in vitro nephron formation from human induced pluripotent stem cells
title_short	PAX2 is dispensable for in vitro nephron formation from human induced pluripotent stem cells
title_full	PAX2 is dispensable for in vitro nephron formation from human induced pluripotent stem cells
title_fullStr	PAX2 is dispensable for in vitro nephron formation from human induced pluripotent stem cells
title_full_unstemmed	PAX2 is dispensable for in vitro nephron formation from human induced pluripotent stem cells
title_sort	pax2 is dispensable for in vitro nephron formation from human induced pluripotent stem cells
publisher	Nature Portfolio
publishDate	2017
url	https://doaj.org/article/5c93a9331aaf4abe88ba9f48b1f8daa1
work_keys_str_mv	AT yusukekaku pax2isdispensableforinvitronephronformationfromhumaninducedpluripotentstemcells AT atsuhirotaguchi pax2isdispensableforinvitronephronformationfromhumaninducedpluripotentstemcells AT shunsuketanigawa pax2isdispensableforinvitronephronformationfromhumaninducedpluripotentstemcells AT fahimhaque pax2isdispensableforinvitronephronformationfromhumaninducedpluripotentstemcells AT tetsushisakuma pax2isdispensableforinvitronephronformationfromhumaninducedpluripotentstemcells AT takashiyamamoto pax2isdispensableforinvitronephronformationfromhumaninducedpluripotentstemcells AT ryuichinishinakamura pax2isdispensableforinvitronephronformationfromhumaninducedpluripotentstemcells
_version_	1718388514255536128

PAX2 is dispensable for in vitro nephron formation from human induced pluripotent stem cells

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