Cyst formation in proximal renal tubules caused by dysfunction of the microtubule minus-end regulator CAMSAP3

Abstract Epithelial cells organize an ordered array of non-centrosomal microtubules, the minus ends of which are regulated by CAMSAP3. The role of these microtubules in epithelial functions, however, is poorly understood. Here, we show that the kidneys of mice in which Camsap3 is mutated develop cys...

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Autores principales:	Yuto Mitsuhata, Takaya Abe, Kazuyo Misaki, Yuna Nakajima, Keita Kiriya, Miwa Kawasaki, Hiroshi Kiyonari, Masatoshi Takeichi, Mika Toya, Masamitsu Sato
Formato:	article
Lenguaje:	EN
Publicado:	Nature Portfolio 2021
Materias:	Medicine R Science Q
Acceso en línea:	https://doaj.org/article/75eea82d1c5a4b1183558e0f3b496943
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spelling	oai:doaj.org-article:75eea82d1c5a4b1183558e0f3b4969432021-12-02T15:52:21ZCyst formation in proximal renal tubules caused by dysfunction of the microtubule minus-end regulator CAMSAP310.1038/s41598-021-85416-x2045-2322https://doaj.org/article/75eea82d1c5a4b1183558e0f3b4969432021-03-01T00:00:00Zhttps://doi.org/10.1038/s41598-021-85416-xhttps://doaj.org/toc/2045-2322Abstract Epithelial cells organize an ordered array of non-centrosomal microtubules, the minus ends of which are regulated by CAMSAP3. The role of these microtubules in epithelial functions, however, is poorly understood. Here, we show that the kidneys of mice in which Camsap3 is mutated develop cysts at the proximal convoluted tubules (PCTs). PCTs were severely dilated in the mutant kidneys, and they also exhibited enhanced cell proliferation. In these PCTs, epithelial cells became flattened along with perturbation of microtubule arrays as well as of certain subcellular structures such as interdigitating basal processes. Furthermore, YAP and PIEZO1, which are known as mechanosensitive regulators for cell shaping and proliferation, were activated in these mutant PCT cells. These observations suggest that CAMSAP3-mediated microtubule networks are important for maintaining the proper mechanical properties of PCT cells, and its loss triggers cell deformation and proliferation via activation of mechanosensors, resulting in the dilation of PCTs.Yuto MitsuhataTakaya AbeKazuyo MisakiYuna NakajimaKeita KiriyaMiwa KawasakiHiroshi KiyonariMasatoshi TakeichiMika ToyaMasamitsu SatoNature PortfolioarticleMedicineRScienceQENScientific Reports, Vol 11, Iss 1, Pp 1-14 (2021)
institution	DOAJ
collection	DOAJ
language	EN
topic	Medicine R Science Q
spellingShingle	Medicine R Science Q Yuto Mitsuhata Takaya Abe Kazuyo Misaki Yuna Nakajima Keita Kiriya Miwa Kawasaki Hiroshi Kiyonari Masatoshi Takeichi Mika Toya Masamitsu Sato Cyst formation in proximal renal tubules caused by dysfunction of the microtubule minus-end regulator CAMSAP3
description	Abstract Epithelial cells organize an ordered array of non-centrosomal microtubules, the minus ends of which are regulated by CAMSAP3. The role of these microtubules in epithelial functions, however, is poorly understood. Here, we show that the kidneys of mice in which Camsap3 is mutated develop cysts at the proximal convoluted tubules (PCTs). PCTs were severely dilated in the mutant kidneys, and they also exhibited enhanced cell proliferation. In these PCTs, epithelial cells became flattened along with perturbation of microtubule arrays as well as of certain subcellular structures such as interdigitating basal processes. Furthermore, YAP and PIEZO1, which are known as mechanosensitive regulators for cell shaping and proliferation, were activated in these mutant PCT cells. These observations suggest that CAMSAP3-mediated microtubule networks are important for maintaining the proper mechanical properties of PCT cells, and its loss triggers cell deformation and proliferation via activation of mechanosensors, resulting in the dilation of PCTs.
format	article
author	Yuto Mitsuhata Takaya Abe Kazuyo Misaki Yuna Nakajima Keita Kiriya Miwa Kawasaki Hiroshi Kiyonari Masatoshi Takeichi Mika Toya Masamitsu Sato
author_facet	Yuto Mitsuhata Takaya Abe Kazuyo Misaki Yuna Nakajima Keita Kiriya Miwa Kawasaki Hiroshi Kiyonari Masatoshi Takeichi Mika Toya Masamitsu Sato
author_sort	Yuto Mitsuhata
title	Cyst formation in proximal renal tubules caused by dysfunction of the microtubule minus-end regulator CAMSAP3
title_short	Cyst formation in proximal renal tubules caused by dysfunction of the microtubule minus-end regulator CAMSAP3
title_full	Cyst formation in proximal renal tubules caused by dysfunction of the microtubule minus-end regulator CAMSAP3
title_fullStr	Cyst formation in proximal renal tubules caused by dysfunction of the microtubule minus-end regulator CAMSAP3
title_full_unstemmed	Cyst formation in proximal renal tubules caused by dysfunction of the microtubule minus-end regulator CAMSAP3
title_sort	cyst formation in proximal renal tubules caused by dysfunction of the microtubule minus-end regulator camsap3
publisher	Nature Portfolio
publishDate	2021
url	https://doaj.org/article/75eea82d1c5a4b1183558e0f3b496943
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Cyst formation in proximal renal tubules caused by dysfunction of the microtubule minus-end regulator CAMSAP3

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