Kidney organoids generated from erythroid progenitors cells of patients with autosomal dominant polycystic kidney disease.

<h4>Background</h4>Kidney organoids have been broadly obtained from commercially available induced pluripotent stem cells (iPSCs); however, it has been a great challenge to efficiently produce renal organoid models from patients with autosomal dominant polycystic kidney disease (ADPKD) t...

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Autores principales: Roberta Facioli, Fernando Henrique Lojudice, Ana Carolina Anauate, Edgar Maquigussa, José Luiz Nishiura, Ita Pfeferman Heilberg, Mari Cleide Sogayar, Mirian Aparecida Boim
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spelling oai:doaj.org-article:faeb29291fd74ba88f7cdc68431437872021-12-02T20:18:54ZKidney organoids generated from erythroid progenitors cells of patients with autosomal dominant polycystic kidney disease.1932-620310.1371/journal.pone.0252156https://doaj.org/article/faeb29291fd74ba88f7cdc68431437872021-01-01T00:00:00Zhttps://doi.org/10.1371/journal.pone.0252156https://doaj.org/toc/1932-6203<h4>Background</h4>Kidney organoids have been broadly obtained from commercially available induced pluripotent stem cells (iPSCs); however, it has been a great challenge to efficiently produce renal organoid models from patients with autosomal dominant polycystic kidney disease (ADPKD) that recapitulate both embryogenesis and the mechanisms of cystogenesis.<h4>Methods</h4>Blood erythroid progenitors (EPs) from two ADPKD patients and one healthy donor (HC) was used as a comparative control to normalize the many technical steps for reprogramming EPs and for the organoids generation. EPs were reprogrammed by an episomal vector into iPSCs, which were differentiated into renal tubular organoids and then stimulated by forskolin to induce cysts formation.<h4>Results</h4>iPSCs derived from EPs exhibited all characteristics of pluripotency and were able to differentiate into all three germ layers. 3D tubular organoids were generated from single cells after 28 days in Matrigel. HC and ADPKD organoids did not spontaneously form cysts, but upon forskolin stimulation, cysts-like structures were observed in the ADPKD organoids but not in the HC-derived organoids.<h4>Conclusion</h4>The findings of this study showed that kidney organoids were successfully generated from the blood EP cells of ADPKD patients and a healthy control donor. This approach should contribute as a powerful tool for embryonic kidney development model, which is able to recapitulate the very early pathophysiological mechanisms involved in cytogenesis.Roberta FacioliFernando Henrique LojudiceAna Carolina AnauateEdgar MaquigussaJosé Luiz NishiuraIta Pfeferman HeilbergMari Cleide SogayarMirian Aparecida BoimPublic Library of Science (PLoS)articleMedicineRScienceQENPLoS ONE, Vol 16, Iss 8, p e0252156 (2021)
institution DOAJ
collection DOAJ
language EN
topic Medicine
R
Science
Q
spellingShingle Medicine
R
Science
Q
Roberta Facioli
Fernando Henrique Lojudice
Ana Carolina Anauate
Edgar Maquigussa
José Luiz Nishiura
Ita Pfeferman Heilberg
Mari Cleide Sogayar
Mirian Aparecida Boim
Kidney organoids generated from erythroid progenitors cells of patients with autosomal dominant polycystic kidney disease.
description <h4>Background</h4>Kidney organoids have been broadly obtained from commercially available induced pluripotent stem cells (iPSCs); however, it has been a great challenge to efficiently produce renal organoid models from patients with autosomal dominant polycystic kidney disease (ADPKD) that recapitulate both embryogenesis and the mechanisms of cystogenesis.<h4>Methods</h4>Blood erythroid progenitors (EPs) from two ADPKD patients and one healthy donor (HC) was used as a comparative control to normalize the many technical steps for reprogramming EPs and for the organoids generation. EPs were reprogrammed by an episomal vector into iPSCs, which were differentiated into renal tubular organoids and then stimulated by forskolin to induce cysts formation.<h4>Results</h4>iPSCs derived from EPs exhibited all characteristics of pluripotency and were able to differentiate into all three germ layers. 3D tubular organoids were generated from single cells after 28 days in Matrigel. HC and ADPKD organoids did not spontaneously form cysts, but upon forskolin stimulation, cysts-like structures were observed in the ADPKD organoids but not in the HC-derived organoids.<h4>Conclusion</h4>The findings of this study showed that kidney organoids were successfully generated from the blood EP cells of ADPKD patients and a healthy control donor. This approach should contribute as a powerful tool for embryonic kidney development model, which is able to recapitulate the very early pathophysiological mechanisms involved in cytogenesis.
format article
author Roberta Facioli
Fernando Henrique Lojudice
Ana Carolina Anauate
Edgar Maquigussa
José Luiz Nishiura
Ita Pfeferman Heilberg
Mari Cleide Sogayar
Mirian Aparecida Boim
author_facet Roberta Facioli
Fernando Henrique Lojudice
Ana Carolina Anauate
Edgar Maquigussa
José Luiz Nishiura
Ita Pfeferman Heilberg
Mari Cleide Sogayar
Mirian Aparecida Boim
author_sort Roberta Facioli
title Kidney organoids generated from erythroid progenitors cells of patients with autosomal dominant polycystic kidney disease.
title_short Kidney organoids generated from erythroid progenitors cells of patients with autosomal dominant polycystic kidney disease.
title_full Kidney organoids generated from erythroid progenitors cells of patients with autosomal dominant polycystic kidney disease.
title_fullStr Kidney organoids generated from erythroid progenitors cells of patients with autosomal dominant polycystic kidney disease.
title_full_unstemmed Kidney organoids generated from erythroid progenitors cells of patients with autosomal dominant polycystic kidney disease.
title_sort kidney organoids generated from erythroid progenitors cells of patients with autosomal dominant polycystic kidney disease.
publisher Public Library of Science (PLoS)
publishDate 2021
url https://doaj.org/article/faeb29291fd74ba88f7cdc6843143787
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