Parkinson's Disease Phenotypes in Patient Neuronal Cultures and Brain Organoids Improved by 2‐Hydroxypropyl‐β‐Cyclodextrin Treatment
ABSTRACT Background The etiology of Parkinson's disease (PD) is only partially understood despite the fact that environmental causes, risk factors, and specific gene mutations are contributors to the disease. Biallelic mutations in the phosphatase and tensin homolog (PTEN)‐induced putative kina...
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| Published in | Movement disorders Vol. 37; no. 1; pp. 80 - 94 |
|---|---|
| Main Authors | , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , |
| Format | Journal Article |
| Language | English |
| Published |
Hoboken, USA
John Wiley & Sons, Inc
01.01.2022
Wiley Subscription Services, Inc |
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| Online Access | Get full text |
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| Abstract | ABSTRACT
Background
The etiology of Parkinson's disease (PD) is only partially understood despite the fact that environmental causes, risk factors, and specific gene mutations are contributors to the disease. Biallelic mutations in the phosphatase and tensin homolog (PTEN)‐induced putative kinase 1 (PINK1) gene involved in mitochondrial homeostasis, vesicle trafficking, and autophagy are sufficient to cause PD.
Objectives
We sought to evaluate the difference between controls' and PINK1 patients' derived neurons in their transition from neuroepithelial stem cells to neurons, allowing us to identify potential pathways to target with repurposed compounds.
Methods
Using two‐dimensional and three‐dimensional models of patients' derived neurons we recapitulated PD‐related phenotypes. We introduced the usage of midbrain organoids for testing compounds. Using Clustered Regularly Interspaced Short Palindromic Repeats (CRISPR)/CRISPR‐associated protein 9 (Cas9), we corrected the point mutations of three patients' derived cells. We evaluated the effect of the selected compound in a mouse model.
Results
PD patient‐derived cells presented differences in their energetic profile, imbalanced proliferation, apoptosis, mitophagy, and a reduced differentiation efficiency to tyrosine hydroxylase positive (TH+) neurons compared to controls' cells. Correction of a patient's point mutation ameliorated the metabolic properties and neuronal firing rates as well as reversing the differentiation phenotype, and reducing the increased astrocytic levels. Treatment with 2‐hydroxypropyl‐β‐cyclodextrin increased the autophagy and mitophagy capacity of neurons concomitant with an improved dopaminergic differentiation of patient‐specific neurons in midbrain organoids and ameliorated neurotoxicity in a mouse model.
Conclusion
We show that treatment with a repurposed compound is sufficient for restoring the impaired dopaminergic differentiation of PD patient‐derived cells. © 2021 The Authors. Movement Disorders published by Wiley Periodicals LLC on behalf of International Parkinson and Movement Disorder Society |
|---|---|
| AbstractList | ABSTRACT
Background
The etiology of Parkinson's disease (PD) is only partially understood despite the fact that environmental causes, risk factors, and specific gene mutations are contributors to the disease. Biallelic mutations in the phosphatase and tensin homolog (PTEN)‐induced putative kinase 1 (
PINK1
) gene involved in mitochondrial homeostasis, vesicle trafficking, and autophagy are sufficient to cause PD.
Objectives
We sought to evaluate the difference between controls' and
PINK1
patients' derived neurons in their transition from neuroepithelial stem cells to neurons, allowing us to identify potential pathways to target with repurposed compounds.
Methods
Using two‐dimensional and three‐dimensional models of patients' derived neurons we recapitulated PD‐related phenotypes. We introduced the usage of midbrain organoids for testing compounds. Using Clustered Regularly Interspaced Short Palindromic Repeats (CRISPR)/CRISPR‐associated protein 9 (Cas9), we corrected the point mutations of three patients' derived cells. We evaluated the effect of the selected compound in a mouse model.
Results
PD patient‐derived cells presented differences in their energetic profile, imbalanced proliferation, apoptosis, mitophagy, and a reduced differentiation efficiency to tyrosine hydroxylase positive (TH+) neurons compared to controls' cells. Correction of a patient's point mutation ameliorated the metabolic properties and neuronal firing rates as well as reversing the differentiation phenotype, and reducing the increased astrocytic levels. Treatment with 2‐hydroxypropyl‐β‐cyclodextrin increased the autophagy and mitophagy capacity of neurons concomitant with an improved dopaminergic differentiation of patient‐specific neurons in midbrain organoids and ameliorated neurotoxicity in a mouse model.
Conclusion
We show that treatment with a repurposed compound is sufficient for restoring the impaired dopaminergic differentiation of PD patient‐derived cells. © 2021 The Authors.
Movement Disorders
published by Wiley Periodicals LLC on behalf of International Parkinson and Movement Disorder Society The etiology of Parkinson's disease (PD) is only partially understood despite the fact that environmental causes, risk factors, and specific gene mutations are contributors to the disease. Biallelic mutations in the phosphatase and tensin homolog (PTEN)-induced putative kinase 1 (PINK1) gene involved in mitochondrial homeostasis, vesicle trafficking, and autophagy are sufficient to cause PD. We sought to evaluate the difference between controls' and PINK1 patients' derived neurons in their transition from neuroepithelial stem cells to neurons, allowing us to identify potential pathways to target with repurposed compounds. Using two-dimensional and three-dimensional models of patients' derived neurons we recapitulated PD-related phenotypes. We introduced the usage of midbrain organoids for testing compounds. Using Clustered Regularly Interspaced Short Palindromic Repeats (CRISPR)/CRISPR-associated protein 9 (Cas9), we corrected the point mutations of three patients' derived cells. We evaluated the effect of the selected compound in a mouse model. PD patient-derived cells presented differences in their energetic profile, imbalanced proliferation, apoptosis, mitophagy, and a reduced differentiation efficiency to tyrosine hydroxylase positive (TH+) neurons compared to controls' cells. Correction of a patient's point mutation ameliorated the metabolic properties and neuronal firing rates as well as reversing the differentiation phenotype, and reducing the increased astrocytic levels. Treatment with 2-hydroxypropyl-β-cyclodextrin increased the autophagy and mitophagy capacity of neurons concomitant with an improved dopaminergic differentiation of patient-specific neurons in midbrain organoids and ameliorated neurotoxicity in a mouse model. We show that treatment with a repurposed compound is sufficient for restoring the impaired dopaminergic differentiation of PD patient-derived cells. © 2021 The Authors. Movement Disorders published by Wiley Periodicals LLC on behalf of International Parkinson and Movement Disorder Society. BackgroundThe etiology of Parkinson's disease (PD) is only partially understood despite the fact that environmental causes, risk factors, and specific gene mutations are contributors to the disease. Biallelic mutations in the phosphatase and tensin homolog (PTEN)‐induced putative kinase 1 (PINK1) gene involved in mitochondrial homeostasis, vesicle trafficking, and autophagy are sufficient to cause PD.ObjectivesWe sought to evaluate the difference between controls' and PINK1 patients' derived neurons in their transition from neuroepithelial stem cells to neurons, allowing us to identify potential pathways to target with repurposed compounds.MethodsUsing two‐dimensional and three‐dimensional models of patients' derived neurons we recapitulated PD‐related phenotypes. We introduced the usage of midbrain organoids for testing compounds. Using Clustered Regularly Interspaced Short Palindromic Repeats (CRISPR)/CRISPR‐associated protein 9 (Cas9), we corrected the point mutations of three patients' derived cells. We evaluated the effect of the selected compound in a mouse model.ResultsPD patient‐derived cells presented differences in their energetic profile, imbalanced proliferation, apoptosis, mitophagy, and a reduced differentiation efficiency to tyrosine hydroxylase positive (TH+) neurons compared to controls' cells. Correction of a patient's point mutation ameliorated the metabolic properties and neuronal firing rates as well as reversing the differentiation phenotype, and reducing the increased astrocytic levels. Treatment with 2‐hydroxypropyl‐β‐cyclodextrin increased the autophagy and mitophagy capacity of neurons concomitant with an improved dopaminergic differentiation of patient‐specific neurons in midbrain organoids and ameliorated neurotoxicity in a mouse model.ConclusionWe show that treatment with a repurposed compound is sufficient for restoring the impaired dopaminergic differentiation of PD patient‐derived cells. © 2021 The Authors. Movement Disorders published by Wiley Periodicals LLC on behalf of International Parkinson and Movement Disorder Society ABSTRACT Background The etiology of Parkinson's disease (PD) is only partially understood despite the fact that environmental causes, risk factors, and specific gene mutations are contributors to the disease. Biallelic mutations in the phosphatase and tensin homolog (PTEN)‐induced putative kinase 1 (PINK1) gene involved in mitochondrial homeostasis, vesicle trafficking, and autophagy are sufficient to cause PD. Objectives We sought to evaluate the difference between controls' and PINK1 patients' derived neurons in their transition from neuroepithelial stem cells to neurons, allowing us to identify potential pathways to target with repurposed compounds. Methods Using two‐dimensional and three‐dimensional models of patients' derived neurons we recapitulated PD‐related phenotypes. We introduced the usage of midbrain organoids for testing compounds. Using Clustered Regularly Interspaced Short Palindromic Repeats (CRISPR)/CRISPR‐associated protein 9 (Cas9), we corrected the point mutations of three patients' derived cells. We evaluated the effect of the selected compound in a mouse model. Results PD patient‐derived cells presented differences in their energetic profile, imbalanced proliferation, apoptosis, mitophagy, and a reduced differentiation efficiency to tyrosine hydroxylase positive (TH+) neurons compared to controls' cells. Correction of a patient's point mutation ameliorated the metabolic properties and neuronal firing rates as well as reversing the differentiation phenotype, and reducing the increased astrocytic levels. Treatment with 2‐hydroxypropyl‐β‐cyclodextrin increased the autophagy and mitophagy capacity of neurons concomitant with an improved dopaminergic differentiation of patient‐specific neurons in midbrain organoids and ameliorated neurotoxicity in a mouse model. Conclusion We show that treatment with a repurposed compound is sufficient for restoring the impaired dopaminergic differentiation of PD patient‐derived cells. © 2021 The Authors. Movement Disorders published by Wiley Periodicals LLC on behalf of International Parkinson and Movement Disorder Society |
| Author | Boussaad, Ibrahim Schmidt, Ronny Schröder, Christoph Brunelli, Francesco Skwirblies, Florian Vitali, Armelle Rakovic, Aleksandar Su, Jihui Li, Wen Schwamborn, Jens C. Gomez‐Giro, Gemma Yuan, Lin Jäger, Christian Klein, Christine Antony, Paul M.A. Griesbeck, Anne Zaffaroni, Gaia Jarazo, Javier Rosety, Isabel Smits, Lisa M. Sabaté‐Soler, Sònia Monzel, Anna S. Qing, Xiaobing Arias‐Fuenzalida, Jonathan Bolognin, Silvia Saraiva, Cláudia Modamio, Jennifer Arena, Giuseppe Krüger, Rejko Berger, Emanuel Walter, Jonas Barmpa, Kyriaki Cruciani, Gerald Seibler, Philip |
| AuthorAffiliation | 12 Transversal Translational Medicine Luxembourg Institute of Health Strassen Luxembourg 2 OrganoTherapeutics société à responsabilité limitée simplifiée (SARL‐S) Esch‐sur‐Alzette Luxembourg 6 Translational Neuroscience, Luxembourg Centre for Systems Biomedicine University of Luxembourg Esch‐sur‐Alzette Luxembourg 1 Developmental and Cellular Biology Luxembourg Centre for Systems Biomedicine University of Luxembourg Esch‐sur‐Alzette Luxembourg 4 Institute for Globally Distributed Open Research and Education Gothenburg Sweden 10 Institute of Neurogenetics University of Lübeck Lübeck Germany 5 Institute of Health Sciences China Medical University Shenyang China 3 Sciomics GmbH Heidelberg Germany 7 Disease Modeling and Screening Platform Luxembourg Institute of Systems Biomedicine, University of Luxembourg and Luxembourg Institute of Health Belvaux Luxembourg 9 Metabolomics Platform, Enzymology and Metabolism Luxembourg Centre for Systems Biomedicine University of Luxembourg Esch‐sur‐Alzette Luxem |
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Jonas orcidid: 0000-0002-9618-0928 surname: Walter fullname: Walter, Jonas organization: Luxembourg Centre for Systems Biomedicine University of Luxembourg – sequence: 14 givenname: Gemma orcidid: 0000-0002-7259-5002 surname: Gomez‐Giro fullname: Gomez‐Giro, Gemma organization: Luxembourg Centre for Systems Biomedicine University of Luxembourg – sequence: 15 givenname: Anna S. orcidid: 0000-0001-8687-6019 surname: Monzel fullname: Monzel, Anna S. organization: Luxembourg Centre for Systems Biomedicine University of Luxembourg – sequence: 16 givenname: Xiaobing surname: Qing fullname: Qing, Xiaobing organization: Luxembourg Centre for Systems Biomedicine University of Luxembourg – sequence: 17 givenname: Armelle surname: Vitali fullname: Vitali, Armelle organization: Translational Neuroscience, Luxembourg Centre for Systems Biomedicine University of Luxembourg – sequence: 18 givenname: Gerald surname: Cruciani fullname: Cruciani, Gerald organization: Luxembourg Institute of Systems Biomedicine, University of Luxembourg and Luxembourg Institute of Health – sequence: 19 givenname: Ibrahim orcidid: 0000-0002-3512-3364 surname: Boussaad fullname: Boussaad, Ibrahim organization: Luxembourg Institute of Systems Biomedicine, University of Luxembourg and Luxembourg Institute of Health – sequence: 20 givenname: Francesco orcidid: 0000-0003-3159-051X surname: Brunelli fullname: Brunelli, Francesco organization: University of Pavia – sequence: 21 givenname: Christian orcidid: 0000-0003-4235-7430 surname: Jäger fullname: Jäger, Christian organization: Luxembourg Centre for Systems Biomedicine University of Luxembourg – sequence: 22 givenname: Aleksandar orcidid: 0000-0002-8127-7911 surname: Rakovic fullname: Rakovic, Aleksandar organization: University of Lübeck – sequence: 23 givenname: Wen surname: Li fullname: Li, Wen organization: China Medical University – sequence: 24 givenname: Lin surname: Yuan fullname: Yuan, Lin organization: China Medical University – sequence: 25 givenname: Emanuel surname: Berger fullname: Berger, Emanuel organization: Luxembourg Centre for Systems Biomedicine University of Luxembourg – sequence: 26 givenname: Giuseppe orcidid: 0000-0003-2398-5503 surname: Arena fullname: Arena, Giuseppe organization: Translational Neuroscience, Luxembourg Centre for Systems Biomedicine University of Luxembourg – sequence: 27 givenname: Silvia orcidid: 0000-0002-1399-2999 surname: Bolognin fullname: Bolognin, Silvia organization: Luxembourg Centre for Systems Biomedicine University of Luxembourg – sequence: 28 givenname: Ronny surname: Schmidt fullname: Schmidt, Ronny organization: Sciomics GmbH – sequence: 29 givenname: Christoph orcidid: 0000-0001-6177-1042 surname: Schröder fullname: Schröder, Christoph organization: Sciomics GmbH – sequence: 30 givenname: Paul M.A. orcidid: 0000-0002-0450-9301 surname: Antony fullname: Antony, Paul M.A. organization: Translational Neuroscience, Luxembourg Centre for Systems Biomedicine University of Luxembourg – sequence: 31 givenname: Christine orcidid: 0000-0003-2102-3431 surname: Klein fullname: Klein, Christine organization: University of Lübeck – sequence: 32 givenname: Rejko orcidid: 0000-0003-4258-6241 surname: Krüger fullname: Krüger, Rejko organization: Luxembourg Institute of Health – sequence: 33 givenname: Philip surname: Seibler fullname: Seibler, Philip organization: University of Lübeck – sequence: 34 givenname: Jens C. orcidid: 0000-0003-4496-0559 surname: Schwamborn fullname: Schwamborn, Jens C. email: jens.schwamborn@uni.lu organization: Luxembourg Centre for Systems Biomedicine University of Luxembourg |
| BackLink | https://www.ncbi.nlm.nih.gov/pubmed/34637165$$D View this record in MEDLINE/PubMed |
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| Copyright | 2021 The Authors. published by Wiley Periodicals LLC on behalf of International Parkinson and Movement Disorder Society 2021 The Authors. Movement Disorders published by Wiley Periodicals LLC on behalf of International Parkinson and Movement Disorder Society. 2021. This article is published under http://creativecommons.org/licenses/by/4.0/ (the “License”). Notwithstanding the ProQuest Terms and Conditions, you may use this content in accordance with the terms of the License. |
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| ISSN | 0885-3185 |
| IngestDate | Tue Sep 17 20:47:14 EDT 2024 Fri Sep 13 02:55:31 EDT 2024 Fri Aug 23 03:27:44 EDT 2024 Thu May 23 23:36:29 EDT 2024 Sat Aug 24 01:09:06 EDT 2024 |
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| Issue | 1 |
| Keywords | PINK1 Parkinson's disease isogenics organoids cyclodextrin |
| Language | English |
| License | Attribution 2021 The Authors. Movement Disorders published by Wiley Periodicals LLC on behalf of International Parkinson and Movement Disorder Society. This is an open access article under the terms of the http://creativecommons.org/licenses/by/4.0/ License, which permits use, distribution and reproduction in any medium, provided the original work is properly cited. |
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| Notes | This project was funded by the Fonds National de la Recherche (FNR) Luxembourg (CORE, C13/BM/5791363). This is a European Union Joint Program–Neurodegenerative Disease Research (JPND) project (INTER/JPND/14/02; INTER/JPND/15/11092422). This project is also supported by the European Union's Horizon 2020 research and innovation program under grant agreement no. 668738, Systems Medicine of Mitochondrial Parkinson's Disease (SysMedPD). The automated screening platform was supported by a PEARL grant of the FNR to R.K. (FNR/P13/6682797). G.A. is supported by the FNR Mitochondrial Risk factors in Parkinson's Disease (MiRisk‐PD) (C17/BM/11676395). J.J. is supported by a Pelican award from the Fondation du Pelican de Mie et Pierre Hippert‐Faber. J.J., L.M.S., A.S.M., J.W., and X.Q. were supported by FNR Aides à la Formation‐Recherche. G.G.G. was funded by the Neuronal ceroid lipofuscinosis (NCL)‐Stiftung (Hamburg, Germany). A.R., C.K., and P.S. are supported by the Deutsche Forschungs Gemeinschaft (DFG) (FOR2488). The copyright line for this article was changed on 27 October 2021, after original online publication. J.J., S.B., and J.C.S. are cofounders and shareholders of OrganoTherapeutics société à responsabilité limitée simplifiée (SARL‐S). C.S. is founder of Sciomics GmbH. Relevant conflicts of interest/financial disclosures Funding agencies Relevant conflicts of interest/financial disclosures: J.J., S.B., and J.C.S. are cofounders and shareholders of OrganoTherapeutics société à responsabilité limitée simplifiée (SARL‐S). C.S. is founder of Sciomics GmbH. Funding agencies: This project was funded by the Fonds National de la Recherche (FNR) Luxembourg (CORE, C13/BM/5791363). This is a European Union Joint Program–Neurodegenerative Disease Research (JPND) project (INTER/JPND/14/02; INTER/JPND/15/11092422). This project is also supported by the European Union's Horizon 2020 research and innovation program under grant agreement no. 668738, Systems Medicine of Mitochondrial Parkinson's Disease (SysMedPD). The automated screening platform was supported by a PEARL grant of the FNR to R.K. (FNR/P13/6682797). G.A. is supported by the FNR Mitochondrial Risk factors in Parkinson's Disease (MiRisk‐PD) (C17/BM/11676395). J.J. is supported by a Pelican award from the Fondation du Pelican de Mie et Pierre Hippert‐Faber. J.J., L.M.S., A.S.M., J.W., and X.Q. were supported by FNR Aides à la Formation‐Recherche. G.G.G. was funded by the Neuronal ceroid lipofuscinosis (NCL)‐Stiftung (Hamburg, Germany). A.R., C.K., and P.S. are supported by the Deutsche Forschungs Gemeinschaft (DFG) (FOR2488). |
| ORCID | 0000-0002-1020-861X 0000-0003-4235-7430 0000-0003-3938-4745 0000-0001-9652-3620 0000-0003-4866-8790 0000-0001-8687-6019 0000-0003-2398-5503 0000-0002-7259-5002 0000-0002-3512-3364 0000-0002-8127-7911 0000-0001-9118-6995 0000-0001-6430-6357 0000-0002-1399-2999 0000-0003-4258-6241 0000-0003-4496-0559 0000-0002-0450-9301 0000-0001-6177-1042 0000-0003-2102-3431 0000-0002-9618-0928 0000-0002-8126-0490 0000-0003-3159-051X |
| OpenAccessLink | https://proxy.k.utb.cz/login?url=https://onlinelibrary.wiley.com/doi/abs/10.1002%2Fmds.28810 |
| PMID | 34637165 |
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| PublicationCentury | 2000 |
| PublicationDate | January 2022 |
| PublicationDateYYYYMMDD | 2022-01-01 |
| PublicationDate_xml | – month: 01 year: 2022 text: January 2022 |
| PublicationDecade | 2020 |
| PublicationPlace | Hoboken, USA |
| PublicationPlace_xml | – name: Hoboken, USA – name: United States – name: Hoboken |
| PublicationTitle | Movement disorders |
| PublicationTitleAlternate | Mov Disord |
| PublicationYear | 2022 |
| Publisher | John Wiley & Sons, Inc Wiley Subscription Services, Inc |
| Publisher_xml | – name: John Wiley & Sons, Inc – name: Wiley Subscription Services, Inc |
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Background
The etiology of Parkinson's disease (PD) is only partially understood despite the fact that environmental causes, risk factors, and... The etiology of Parkinson's disease (PD) is only partially understood despite the fact that environmental causes, risk factors, and specific gene mutations are... BackgroundThe etiology of Parkinson's disease (PD) is only partially understood despite the fact that environmental causes, risk factors, and specific gene... |
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| SubjectTerms | 2-Hydroxypropyl-beta-cyclodextrin - metabolism Animal models Animals Apoptosis Autophagy Brain - metabolism Cell differentiation Cell proliferation CRISPR cyclodextrin Dopamine receptors Dopaminergic Neurons - metabolism Etiology Homeostasis Humans Hydroxylase isogenics Kinases Mesencephalon Mice Mitochondria Movement disorders Mutation Neurodegenerative diseases Neurons Neurons - metabolism Neurotoxicity organoids Organoids - metabolism Parkinson Disease - drug therapy Parkinson Disease - genetics Parkinson Disease - metabolism Parkinson's disease Patients Phagocytosis Phenotype Phenotypes PINK1 Point mutation PTEN protein PTEN-induced putative kinase Regular Issue Stem cells β-Cyclodextrin |
| Title | Parkinson's Disease Phenotypes in Patient Neuronal Cultures and Brain Organoids Improved by 2‐Hydroxypropyl‐β‐Cyclodextrin Treatment |
| URI | https://onlinelibrary.wiley.com/doi/abs/10.1002%2Fmds.28810 https://www.ncbi.nlm.nih.gov/pubmed/34637165 https://www.proquest.com/docview/2620936296/abstract/ https://pubmed.ncbi.nlm.nih.gov/PMC9291890 |
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