The ciliary gene INPP5E confers dorsal telencephalic identity to human cortical organoids by negatively regulating Sonic Hedgehog signalling

Leah Schembs; Ariane Willems; Kerstin Hasenpusch-Theil; James Cooper; Katie Whiting; Karen Burr; Sunniva Bostrand; Bhuvaneish T. Selvaraj; Siddharthan Chandran; Thomas Theil

doi:10.1016/j.celrep.2022.110811

The ciliary gene INPP5E confers dorsal telencephalic identity to human cortical organoids by negatively regulating Sonic Hedgehog signalling

Leah Schembs, Ariane Willems, Kerstin Hasenpusch-Theil, James Cooper, Katie Whiting, Karen Burr, Sunniva Bostrand, Bhuvaneish T. Selvaraj, Siddharthan Chandran, Thomas Theil

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

Original language	English
Journal	Cell Reports
Early online date	17 May 2022
DOIs	https://doi.org/10.1016/j.celrep.2022.110811
Publication status	E-pub ahead of print - 17 May 2022

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10.1016/j.celrep.2022.110811

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Schembs, L., Willems, A., Hasenpusch-Theil, K., Cooper, J., Whiting, K., Burr, K., Bostrand, S., Selvaraj, B. T., Chandran, S., & Theil, T. (2022). The ciliary gene INPP5E confers dorsal telencephalic identity to human cortical organoids by negatively regulating Sonic Hedgehog signalling. Cell Reports. Advance online publication. https://doi.org/10.1016/j.celrep.2022.110811

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title = "The ciliary gene INPP5E confers dorsal telencephalic identity to human cortical organoids by negatively regulating Sonic Hedgehog signalling",

author = "Leah Schembs and Ariane Willems and Kerstin Hasenpusch-Theil and James Cooper and Katie Whiting and Karen Burr and Sunniva Bostrand and Selvaraj, {Bhuvaneish T.} and Siddharthan Chandran and Thomas Theil",

note = "Funding Information: We are grateful to Drs. Thomas Becker, John Mason, Pleasantine Mill, and David Price for critical comments on the manuscript and Owen Dando for advice on bioinformatics. The human embryonic and fetal material was provided by the Joint MRC / Wellcome Trust (grant no. MR/R006237/1 ) Human Developmental Biology Resource ( www.hdbr.org ). This work was supported by grants from an RS Macdonald Seedcord fund and from the Simons Initiative for the Developing Brain (SFARI - 529085 ) to T.T. and S.C. The Chandran lab is supported by the Euan MacDonald Center for Motor Neurone Disease Research and the UK Dementia Research Institute (DRI), which receives its funding from UK DRI Ltd., funded by the UK Medical Research Council , Alzheimer{\textquoteright}s Society, and Alzheimer{\textquoteright}s Research UK (217CHA R83786 (MRC) = DRI-CORE-2017-EDI). B.T.S. is a Rowling/ DRI fellow, funded by the “ Anne Rowling Regenerative Neurology Clinic .” S.M.K.B. is funded by the Wellcome Trust Translational Neuroscience PhD Programme at the University of Edinburgh ( 108890/Z/15/Z ). We also acknowledge the financial support of NHS Research Scotland (NRS), through Edinburgh Clinical Research Facility. Funding Information: We are grateful to Drs. Thomas Becker, John Mason, Pleasantine Mill, and David Price for critical comments on the manuscript and Owen Dando for advice on bioinformatics. The human embryonic and fetal material was provided by the Joint MRC/Wellcome Trust (grant no. MR/R006237/1) Human Developmental Biology Resource (www.hdbr.org). This work was supported by grants from an RS Macdonald Seedcord fund and from the Simons Initiative for the Developing Brain (SFARI -529085) to T.T. and S.C. The Chandran lab is supported by the Euan MacDonald Center for Motor Neurone Disease Research and the UK Dementia Research Institute (DRI), which receives its funding from UK DRI Ltd. funded by the UK Medical Research Council, Alzheimer's Society, and Alzheimer's Research UK (217CHA R83786 (MRC) = DRI-CORE-2017-EDI). B.T.S. is a Rowling/DRI fellow, funded by the “Anne Rowling Regenerative Neurology Clinic.” S.M.K.B. is funded by the Wellcome Trust Translational Neuroscience PhD Programme at the University of Edinburgh (108890/Z/15/Z). We also acknowledge the financial support of NHS Research Scotland (NRS), through Edinburgh Clinical Research Facility. Conceptualization, B.T.S. S.C. and T.T.; investigation, L.S. A.W. K.H.-T. J.D.C. K.W. K.B. S.M.K.B. B.T.S. and T.T.; writing, A.W. B.T.S. and T.T. The authors declare no competing interests. One or more of the authors of this paper self-identifies as a member of the LGBTQ+ community. Publisher Copyright: {\textcopyright} 2022 The Authors",

year = "2022",

month = may,

day = "17",

doi = "10.1016/j.celrep.2022.110811",

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T1 - The ciliary gene INPP5E confers dorsal telencephalic identity to human cortical organoids by negatively regulating Sonic Hedgehog signalling

AU - Schembs, Leah

AU - Willems, Ariane

AU - Hasenpusch-Theil, Kerstin

AU - Cooper, James

AU - Whiting, Katie

AU - Burr, Karen

AU - Bostrand, Sunniva

AU - Selvaraj, Bhuvaneish T.

AU - Chandran, Siddharthan

AU - Theil, Thomas

N1 - Funding Information: We are grateful to Drs. Thomas Becker, John Mason, Pleasantine Mill, and David Price for critical comments on the manuscript and Owen Dando for advice on bioinformatics. The human embryonic and fetal material was provided by the Joint MRC / Wellcome Trust (grant no. MR/R006237/1 ) Human Developmental Biology Resource ( www.hdbr.org ). This work was supported by grants from an RS Macdonald Seedcord fund and from the Simons Initiative for the Developing Brain (SFARI - 529085 ) to T.T. and S.C. The Chandran lab is supported by the Euan MacDonald Center for Motor Neurone Disease Research and the UK Dementia Research Institute (DRI), which receives its funding from UK DRI Ltd., funded by the UK Medical Research Council , Alzheimer’s Society, and Alzheimer’s Research UK (217CHA R83786 (MRC) = DRI-CORE-2017-EDI). B.T.S. is a Rowling/ DRI fellow, funded by the “ Anne Rowling Regenerative Neurology Clinic .” S.M.K.B. is funded by the Wellcome Trust Translational Neuroscience PhD Programme at the University of Edinburgh ( 108890/Z/15/Z ). We also acknowledge the financial support of NHS Research Scotland (NRS), through Edinburgh Clinical Research Facility. Funding Information: We are grateful to Drs. Thomas Becker, John Mason, Pleasantine Mill, and David Price for critical comments on the manuscript and Owen Dando for advice on bioinformatics. The human embryonic and fetal material was provided by the Joint MRC/Wellcome Trust (grant no. MR/R006237/1) Human Developmental Biology Resource (www.hdbr.org). This work was supported by grants from an RS Macdonald Seedcord fund and from the Simons Initiative for the Developing Brain (SFARI -529085) to T.T. and S.C. The Chandran lab is supported by the Euan MacDonald Center for Motor Neurone Disease Research and the UK Dementia Research Institute (DRI), which receives its funding from UK DRI Ltd. funded by the UK Medical Research Council, Alzheimer's Society, and Alzheimer's Research UK (217CHA R83786 (MRC) = DRI-CORE-2017-EDI). B.T.S. is a Rowling/DRI fellow, funded by the “Anne Rowling Regenerative Neurology Clinic.” S.M.K.B. is funded by the Wellcome Trust Translational Neuroscience PhD Programme at the University of Edinburgh (108890/Z/15/Z). We also acknowledge the financial support of NHS Research Scotland (NRS), through Edinburgh Clinical Research Facility. Conceptualization, B.T.S. S.C. and T.T.; investigation, L.S. A.W. K.H.-T. J.D.C. K.W. K.B. S.M.K.B. B.T.S. and T.T.; writing, A.W. B.T.S. and T.T. The authors declare no competing interests. One or more of the authors of this paper self-identifies as a member of the LGBTQ+ community. Publisher Copyright: © 2022 The Authors

PY - 2022/5/17

Y1 - 2022/5/17

U2 - 10.1016/j.celrep.2022.110811

DO - 10.1016/j.celrep.2022.110811

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SN - 2211-1247

JO - Cell Reports

JF - Cell Reports

ER -