Literature DB >> 32058042

Deep phenotyping of peripheral tissue facilitates mechanistic disease stratification in sporadic Parkinson's disease.

Phillippa J Carling1, Heather Mortiboys1, Claire Green1, Simeon Mihaylov1, Cynthia Sandor2, Aurelie Schwartzentruber1, Rosie Taylor3, Wenbin Wei1, Chris Hastings1, Siew Wong1, Christine Lo1, Samuel Evetts4, Hannah Clemmens1, Matthew Wyles1, Sam Willcox1, Thomas Payne1, Rachel Hughes1, Laura Ferraiuolo1, Caleb Webber5, Winston Hide6, Richard Wade-Martins7, Kevin Talbot8, Michele T Hu4, Oliver Bandmann9.   

Abstract

Mechanistic disease stratification will be crucial to develop a precision medicine approach for future disease modifying therapy in sporadic Parkinson's disease (sPD). Mitochondrial and lysosomal dysfunction are key mechanisms in the pathogenesis of sPD and therefore promising targets for therapeutic intervention. We investigated mitochondrial and lysosomal function in skin fibroblasts of 100 sPD patients and 50 age-matched controls. A combination of cellular assays, RNA-seq based pathway analysis and genotyping was applied. Distinct subgroups with mitochondrial (mito-sPD) or lysosomal (lyso-sPD) dysfunction were identified. Mitochondrial dysfunction correlated with reduction in complex I and IV protein levels. RNA-seq based pathway analysis revealed marked activation of the lysosomal pathway with enrichment for lysosomal disease gene variants in lyso-sPD. Conversion of fibroblasts to induced neuronal progenitor cells and subsequent differentiation into tyrosine hydroxylase positive neurons confirmed and further enhanced both mitochondrial and lysosomal abnormalities. Treatment with ursodeoxycholic acid improved mitochondrial membrane potential and intracellular ATP levels even in sPD patient fibroblast lines with comparatively mild mitochondrial dysfunction. The results of our study suggest that in-depth phenotyping and focussed assessment of putative neuroprotective compounds in peripheral tissue are a promising approach towards disease stratification and precision medicine in sPD.
Copyright © 2020. Published by Elsevier Ltd.

Entities:  

Keywords:  Disease stratification; Fibroblasts; Parkinson’s disease; UDCA; Ursodeoxycholic acid

Year:  2020        PMID: 32058042     DOI: 10.1016/j.pneurobio.2020.101772

Source DB:  PubMed          Journal:  Prog Neurobiol        ISSN: 0301-0082            Impact factor:   11.685


  10 in total

1.  Primary cilia and SHH signaling impairments in human and mouse models of Parkinson's disease.

Authors:  Sebastian Schmidt; Malte D Luecken; Dietrich Trümbach; Sina Hembach; Kristina M Niedermeier; Nicole Wenck; Klaus Pflügler; Constantin Stautner; Anika Böttcher; Heiko Lickert; Ciro Ramirez-Suastegui; Ruhel Ahmad; Michael J Ziller; Julia C Fitzgerald; Viktoria Ruf; Wilma D J van de Berg; Allert J Jonker; Thomas Gasser; Beate Winner; Jürgen Winkler; Daniela M Vogt Weisenhorn; Florian Giesert; Fabian J Theis; Wolfgang Wurst
Journal:  Nat Commun       Date:  2022-08-16       Impact factor: 17.694

2.  Analysis of hemisphere-dependent effects of unilateral intrastriatal injection of α-synuclein pre-formed fibrils on mitochondrial protein levels, dynamics, and function.

Authors:  Rose B Creed; Adeel A Memon; Sindhu P Komaragiri; Sandeep K Barodia; Matthew S Goldberg
Journal:  Acta Neuropathol Commun       Date:  2022-05-23       Impact factor: 7.578

3.  Deficits in Mitochondrial Spare Respiratory Capacity Contribute to the Neuropsychological Changes of Alzheimer's Disease.

Authors:  Simon M Bell; Matteo De Marco; Katy Barnes; Pamela J Shaw; Laura Ferraiuolo; Daniel J Blackburn; Heather Mortiboys; Annalena Venneri
Journal:  J Pers Med       Date:  2020-04-29

Review 4.  Drug Repurposing for Parkinson's Disease: The International Linked Clinical Trials experience.

Authors:  Simon R W Stott; Richard K Wyse; Patrik Brundin
Journal:  Front Neurosci       Date:  2021-03-19       Impact factor: 4.677

5.  Benchmarking a highly selective USP30 inhibitor for enhancement of mitophagy and pexophagy.

Authors:  Emma V Rusilowicz-Jones; Francesco G Barone; Fernanda Martins Lopes; Elezabeth Stephen; Heather Mortiboys; Sylvie Urbé; Michael J Clague
Journal:  Life Sci Alliance       Date:  2021-11-29

6.  Adult health and transition stage-specific rotenone-mediated Drosophila model of Parkinson's disease: Impact on late-onset neurodegenerative disease models.

Authors:  Mohamad Ayajuddin; Limamanen Phom; Zevelou Koza; Priyanka Modi; Abhik Das; Rahul Chaurasia; Abuno Thepa; Nukshimenla Jamir; Kelevikho Neikha; Sarat Chandra Yenisetti
Journal:  Front Mol Neurosci       Date:  2022-08-09       Impact factor: 6.261

7.  Striatal synaptic bioenergetic and autophagic decline in premotor experimental parkinsonism.

Authors:  Leyre Merino-Galán; Haritz Jimenez-Urbieta; Marta Zamarbide; Tatiana Rodríguez-Chinchilla; Arantzazu Belloso-Iguerategui; Enrique Santamaria; Joaquín Fernández-Irigoyen; Ana Aiastui; Evelyne Doudnikoff; Erwan Bézard; Alberto Ouro; Shira Knafo; Belén Gago; Ana Quiroga-Varela; María Cruz Rodríguez-Oroz
Journal:  Brain       Date:  2022-06-30       Impact factor: 15.255

8.  Oxidative switch drives mitophagy defects in dopaminergic parkin mutant patient neurons.

Authors:  Aurelie Schwartzentruber; Camilla Boschian; Fernanda Martins Lopes; Monika A Myszczynska; Elizabeth J New; Julien Beyrath; Jan Smeitink; Laura Ferraiuolo; Heather Mortiboys
Journal:  Sci Rep       Date:  2020-09-23       Impact factor: 4.379

Review 9.  Omics Data and Their Integrative Analysis to Support Stratified Medicine in Neurodegenerative Diseases.

Authors:  Valentina La Cognata; Giovanna Morello; Sebastiano Cavallaro
Journal:  Int J Mol Sci       Date:  2021-05-01       Impact factor: 5.923

Review 10.  Is there a special relationship between complex I activity and nigral neuronal loss in Parkinson's disease? A critical reappraisal.

Authors:  Nitya Subrahmanian; Matthew J LaVoie
Journal:  Brain Res       Date:  2021-03-19       Impact factor: 3.610

  10 in total

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