Human pluripotent stem cell-derived kidney organoids for personalized congenital and idiopathic nephrotic syndrome modeling

J. Jansen; B. T. van den Berge; M. van den Broek; R. J. Maas; D. Daviran; B. Willemsen; R. Roverts; M. van der Kruit; C. Kuppe; K. C. Reimer; G. D. Giovanni; F. Mooren; Q. Nlandu; H. Mudde; R. Wetzels; D. den Braanker; N. Parr; J. S. Nagai; V. Drenic; I. G. Costa; E. Steenbergen; T. Nijenhuis; H. Dijkman; N. Endlich; N. C. A. J. van de Kar; R. K. Schneider; J. F. M. Wetzels; A. Akiva; J. van der Vlag; R. Kramann; M. F. Schreuder; B. Smeets

doi:10.1242/dev.200198

Human pluripotent stem cell-derived kidney organoids for personalized congenital and idiopathic nephrotic syndrome modeling

J. Jansen^*, B. T. van den Berge, M. van den Broek, R. J. Maas, D. Daviran, B. Willemsen, R. Roverts, M. van der Kruit, C. Kuppe, K. C. Reimer, G. D. Giovanni, F. Mooren, Q. Nlandu, H. Mudde, R. Wetzels, D. den Braanker, N. Parr, J. S. Nagai, V. Drenic, I. G. CostaE. Steenbergen, T. Nijenhuis, H. Dijkman, N. Endlich, N. C. A. J. van de Kar, R. K. Schneider, J. F. M. Wetzels, A. Akiva, J. van der Vlag, R. Kramann, M. F. Schreuder, B. Smeets

^*Corresponding author for this work

Research output: Contribution to journal › Article › Academic › peer-review

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Abstract

Nephrotic syndrome (NS) is characterized by severe proteinuria as a consequence of kidney glomerular injury due to podocyte damage. In vitro models mimicking in vivo podocyte characteristics are a prerequisite to resolve NS pathogenesis.

Original language	English
Article number	dev200198
Journal	Development (Cambridge)
Volume	149
Issue number	9
DOIs	https://doi.org/10.1242/dev.200198
Publication status	Published - 6 May 2022

Bibliographical note

Funding Information:
We would like to thank the Stem Cell Technology Center, Radboudumc, Nijmegen, The Netherlands, and the iPS Core Facility, Erasmus Medical Center, Rotterdam, The Netherlands, for generating iPSCs and performing the gene editing. We would like to thank Nihal Telli for her help in the super-resolution filtration slit imaging. We would like to thank the Radboudumc Technology Centers bioinformatics, microscopy and flow cytometry for their support. We thank Mariska Kea-te Lindert for her help in sample preparation for electron microscopy. J.J. is supported by The Netherlands Organization for Scientific Research (Nederlandse Organisatie voor Wetenschappelijk Onderzoek; NWO Veni grant 091 501 61 81 01 36) and the Dutch Kidney Foundation (Nierstichting; 19OK005). B.T.v.d.B. is supported by grants awarded to R.J.M.: the Netherlands Organization for Scientific Research (NWO Off road grant 451001034) and the Dutch Kidney Foundation (18OKG05 and 18OI14). M.v.d.B. is supported by the Dutch Kidney Foundation (19OK005). D.D., R.R. and A.A. are supported by a European Research Council Advanced Investigator grant (H2020-ERC-2017-ADV-788982-COLMIN) to Nico Sommerdijk. A.A. is also supported by the NWO (VI.Veni.192.094). M.F.S. is supported by the Dutch Kidney Foundation (15OKG16) and The Netherlands Organization for Scientific Research (NWO VIDI grant 016.156.454). B.S. is supported by the Dutch Kidney Foundation (14A3D104) and The Netherlands Organization for Scientific Research (NWO VIDI grant 016.156.363). J.S.N. and I.G.C. are supported by the E:MED Consortia Fibromap funded by the German Ministry of Education and Science (Bundesministerium für Bildung und Forschung). Open Access funding provided by Radboud University: Radboud Universiteit. Deposited in PMC for immediate release.

Funding Information:
J.J. is supported by The Netherlands Organization for Scientific Research (Nederlandse Organisatie voor Wetenschappelijk Onderzoek; NWO Veni grant 091 501 61 81 01 36) and the Dutch Kidney Foundation (Nierstichting; 19OK005). B.T.v.d.B. is supported by grants awarded to R.J.M.: the Netherlands Organization for Scientific Research (NWO Off road grant 451001034) and the Dutch Kidney Foundation (18OKG05 and 18OI14). M.v.d.B. is supported by the Dutch Kidney Foundation (19OK005). D.D., R.R. and A.A. are supported by a European Research Council Advanced Investigator grant (H2020-ERC-2017-ADV-788982-COLMIN) to Nico Sommerdijk. A.A. is also supported by the NWO (VI.Veni.192.094). M.F.S. is supported by the Dutch Kidney Foundation (15OKG16) and The Netherlands Organization for Scientific Research (NWO VIDI grant 016.156.454). B.S. is supported by the Dutch Kidney Foundation (14A3D104) and The Netherlands Organization for Scientific Research (NWO VIDI grant 016.156.363). J.S.N. and I.G.C. are supported by the E:MED Consortia Fibromap funded by the German Ministry of Education and Science (Bundesministerium für Bildung und Forschung). Open Access funding provided by Radboud University: Radboud Universiteit. Deposited in PMC for immediate release.

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© 2022. Published by The Company of Biologists Ltd

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Jansen, J., van den Berge, B. T., van den Broek, M., Maas, R. J., Daviran, D., Willemsen, B., Roverts, R., van der Kruit, M., Kuppe, C., Reimer, K. C., Giovanni, G. D., Mooren, F., Nlandu, Q., Mudde, H., Wetzels, R., den Braanker, D., Parr, N., Nagai, J. S., Drenic, V., ... Smeets, B. (2022). Human pluripotent stem cell-derived kidney organoids for personalized congenital and idiopathic nephrotic syndrome modeling. Development (Cambridge), 149(9), Article dev200198. https://doi.org/10.1242/dev.200198

@article{338c3cafc94c48969c0faa159c09dd6b,

title = "Human pluripotent stem cell-derived kidney organoids for personalized congenital and idiopathic nephrotic syndrome modeling",

abstract = "Nephrotic syndrome (NS) is characterized by severe proteinuria as a consequence of kidney glomerular injury due to podocyte damage. In vitro models mimicking in vivo podocyte characteristics are a prerequisite to resolve NS pathogenesis.",

author = "J. Jansen and {van den Berge}, {B. T.} and {van den Broek}, M. and Maas, {R. J.} and D. Daviran and B. Willemsen and R. Roverts and {van der Kruit}, M. and C. Kuppe and Reimer, {K. C.} and Giovanni, {G. D.} and F. Mooren and Q. Nlandu and H. Mudde and R. Wetzels and {den Braanker}, D. and N. Parr and Nagai, {J. S.} and V. Drenic and Costa, {I. G.} and E. Steenbergen and T. Nijenhuis and H. Dijkman and N. Endlich and {van de Kar}, {N. C. A. J.} and Schneider, {R. K.} and Wetzels, {J. F. M.} and A. Akiva and {van der Vlag}, J. and R. Kramann and Schreuder, {M. F.} and B. Smeets",

note = "Funding Information: We would like to thank the Stem Cell Technology Center, Radboudumc, Nijmegen, The Netherlands, and the iPS Core Facility, Erasmus Medical Center, Rotterdam, The Netherlands, for generating iPSCs and performing the gene editing. We would like to thank Nihal Telli for her help in the super-resolution filtration slit imaging. We would like to thank the Radboudumc Technology Centers bioinformatics, microscopy and flow cytometry for their support. We thank Mariska Kea-te Lindert for her help in sample preparation for electron microscopy. J.J. is supported by The Netherlands Organization for Scientific Research (Nederlandse Organisatie voor Wetenschappelijk Onderzoek; NWO Veni grant 091 501 61 81 01 36) and the Dutch Kidney Foundation (Nierstichting; 19OK005). B.T.v.d.B. is supported by grants awarded to R.J.M.: the Netherlands Organization for Scientific Research (NWO Off road grant 451001034) and the Dutch Kidney Foundation (18OKG05 and 18OI14). M.v.d.B. is supported by the Dutch Kidney Foundation (19OK005). D.D., R.R. and A.A. are supported by a European Research Council Advanced Investigator grant (H2020-ERC-2017-ADV-788982-COLMIN) to Nico Sommerdijk. A.A. is also supported by the NWO (VI.Veni.192.094). M.F.S. is supported by the Dutch Kidney Foundation (15OKG16) and The Netherlands Organization for Scientific Research (NWO VIDI grant 016.156.454). B.S. is supported by the Dutch Kidney Foundation (14A3D104) and The Netherlands Organization for Scientific Research (NWO VIDI grant 016.156.363). J.S.N. and I.G.C. are supported by the E:MED Consortia Fibromap funded by the German Ministry of Education and Science (Bundesministerium f{\"u}r Bildung und Forschung). Open Access funding provided by Radboud University: Radboud Universiteit. Deposited in PMC for immediate release. Funding Information: J.J. is supported by The Netherlands Organization for Scientific Research (Nederlandse Organisatie voor Wetenschappelijk Onderzoek; NWO Veni grant 091 501 61 81 01 36) and the Dutch Kidney Foundation (Nierstichting; 19OK005). B.T.v.d.B. is supported by grants awarded to R.J.M.: the Netherlands Organization for Scientific Research (NWO Off road grant 451001034) and the Dutch Kidney Foundation (18OKG05 and 18OI14). M.v.d.B. is supported by the Dutch Kidney Foundation (19OK005). D.D., R.R. and A.A. are supported by a European Research Council Advanced Investigator grant (H2020-ERC-2017-ADV-788982-COLMIN) to Nico Sommerdijk. A.A. is also supported by the NWO (VI.Veni.192.094). M.F.S. is supported by the Dutch Kidney Foundation (15OKG16) and The Netherlands Organization for Scientific Research (NWO VIDI grant 016.156.454). B.S. is supported by the Dutch Kidney Foundation (14A3D104) and The Netherlands Organization for Scientific Research (NWO VIDI grant 016.156.363). J.S.N. and I.G.C. are supported by the E:MED Consortia Fibromap funded by the German Ministry of Education and Science (Bundesministerium f{\"u}r Bildung und Forschung). Open Access funding provided by Radboud University: Radboud Universiteit. Deposited in PMC for immediate release. Publisher Copyright: {\textcopyright} 2022. Published by The Company of Biologists Ltd",

year = "2022",

month = may,

day = "6",

doi = "10.1242/dev.200198",

language = "English",

volume = "149",

number = "9",

}

Jansen, J, van den Berge, BT, van den Broek, M, Maas, RJ, Daviran, D, Willemsen, B, Roverts, R, van der Kruit, M, Kuppe, C, Reimer, KC, Giovanni, GD, Mooren, F, Nlandu, Q, Mudde, H, Wetzels, R, den Braanker, D, Parr, N, Nagai, JS, Drenic, V, Costa, IG, Steenbergen, E, Nijenhuis, T, Dijkman, H, Endlich, N, van de Kar, NCAJ, Schneider, RK, Wetzels, JFM, Akiva, A, van der Vlag, J, Kramann, R, Schreuder, MF & Smeets, B 2022, 'Human pluripotent stem cell-derived kidney organoids for personalized congenital and idiopathic nephrotic syndrome modeling', Development (Cambridge), vol. 149, no. 9, dev200198. https://doi.org/10.1242/dev.200198

TY - JOUR

T1 - Human pluripotent stem cell-derived kidney organoids for personalized congenital and idiopathic nephrotic syndrome modeling

AU - Jansen, J.

AU - van den Berge, B. T.

AU - van den Broek, M.

AU - Maas, R. J.

AU - Daviran, D.

AU - Willemsen, B.

AU - Roverts, R.

AU - van der Kruit, M.

AU - Kuppe, C.

AU - Reimer, K. C.

AU - Giovanni, G. D.

AU - Mooren, F.

AU - Nlandu, Q.

AU - Mudde, H.

AU - Wetzels, R.

AU - den Braanker, D.

AU - Parr, N.

AU - Nagai, J. S.

AU - Drenic, V.

AU - Costa, I. G.

AU - Steenbergen, E.

AU - Nijenhuis, T.

AU - Dijkman, H.

AU - Endlich, N.

AU - van de Kar, N. C. A. J.

AU - Schneider, R. K.

AU - Wetzels, J. F. M.

AU - Akiva, A.

AU - van der Vlag, J.

AU - Kramann, R.

AU - Schreuder, M. F.

AU - Smeets, B.

N1 - Funding Information: We would like to thank the Stem Cell Technology Center, Radboudumc, Nijmegen, The Netherlands, and the iPS Core Facility, Erasmus Medical Center, Rotterdam, The Netherlands, for generating iPSCs and performing the gene editing. We would like to thank Nihal Telli for her help in the super-resolution filtration slit imaging. We would like to thank the Radboudumc Technology Centers bioinformatics, microscopy and flow cytometry for their support. We thank Mariska Kea-te Lindert for her help in sample preparation for electron microscopy. J.J. is supported by The Netherlands Organization for Scientific Research (Nederlandse Organisatie voor Wetenschappelijk Onderzoek; NWO Veni grant 091 501 61 81 01 36) and the Dutch Kidney Foundation (Nierstichting; 19OK005). B.T.v.d.B. is supported by grants awarded to R.J.M.: the Netherlands Organization for Scientific Research (NWO Off road grant 451001034) and the Dutch Kidney Foundation (18OKG05 and 18OI14). M.v.d.B. is supported by the Dutch Kidney Foundation (19OK005). D.D., R.R. and A.A. are supported by a European Research Council Advanced Investigator grant (H2020-ERC-2017-ADV-788982-COLMIN) to Nico Sommerdijk. A.A. is also supported by the NWO (VI.Veni.192.094). M.F.S. is supported by the Dutch Kidney Foundation (15OKG16) and The Netherlands Organization for Scientific Research (NWO VIDI grant 016.156.454). B.S. is supported by the Dutch Kidney Foundation (14A3D104) and The Netherlands Organization for Scientific Research (NWO VIDI grant 016.156.363). J.S.N. and I.G.C. are supported by the E:MED Consortia Fibromap funded by the German Ministry of Education and Science (Bundesministerium für Bildung und Forschung). Open Access funding provided by Radboud University: Radboud Universiteit. Deposited in PMC for immediate release. Funding Information: J.J. is supported by The Netherlands Organization for Scientific Research (Nederlandse Organisatie voor Wetenschappelijk Onderzoek; NWO Veni grant 091 501 61 81 01 36) and the Dutch Kidney Foundation (Nierstichting; 19OK005). B.T.v.d.B. is supported by grants awarded to R.J.M.: the Netherlands Organization for Scientific Research (NWO Off road grant 451001034) and the Dutch Kidney Foundation (18OKG05 and 18OI14). M.v.d.B. is supported by the Dutch Kidney Foundation (19OK005). D.D., R.R. and A.A. are supported by a European Research Council Advanced Investigator grant (H2020-ERC-2017-ADV-788982-COLMIN) to Nico Sommerdijk. A.A. is also supported by the NWO (VI.Veni.192.094). M.F.S. is supported by the Dutch Kidney Foundation (15OKG16) and The Netherlands Organization for Scientific Research (NWO VIDI grant 016.156.454). B.S. is supported by the Dutch Kidney Foundation (14A3D104) and The Netherlands Organization for Scientific Research (NWO VIDI grant 016.156.363). J.S.N. and I.G.C. are supported by the E:MED Consortia Fibromap funded by the German Ministry of Education and Science (Bundesministerium für Bildung und Forschung). Open Access funding provided by Radboud University: Radboud Universiteit. Deposited in PMC for immediate release. Publisher Copyright: © 2022. Published by The Company of Biologists Ltd

PY - 2022/5/6

Y1 - 2022/5/6

N2 - Nephrotic syndrome (NS) is characterized by severe proteinuria as a consequence of kidney glomerular injury due to podocyte damage. In vitro models mimicking in vivo podocyte characteristics are a prerequisite to resolve NS pathogenesis.

AB - Nephrotic syndrome (NS) is characterized by severe proteinuria as a consequence of kidney glomerular injury due to podocyte damage. In vitro models mimicking in vivo podocyte characteristics are a prerequisite to resolve NS pathogenesis.

UR - http://www.scopus.com/inward/record.url?scp=85129997517&partnerID=8YFLogxK

U2 - 10.1242/dev.200198

DO - 10.1242/dev.200198

M3 - Article

C2 - 35417019

SN - 0950-1991

VL - 149

JO - Development (Cambridge)

JF - Development (Cambridge)

IS - 9

M1 - dev200198

ER -

Human pluripotent stem cell-derived kidney organoids for personalized congenital and idiopathic nephrotic syndrome modeling

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