Drosophila screen connects nuclear transport genes to DPR pathology in c9ALS/FTD

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Items 101–200 of 262

1. Nuclear localized C9orf72-associated arginine-containing dipeptides exhibit age-dependent toxicity in C. elegans
   Paige D. Rudich, Carley Snoznik, Simon C. Watkins, John Monaghan, Udai Bhan Pandey, S. Todd Lamitina · 2017 · Human Molecular Genetics
2. Phasing in on the cell cycle
   Steven Boeynaems, Péter Tompa, Ludo Van Den Bosch · 2018 · Cell Division
3. Symmetric dimethylation of poly-GR correlates with disease duration in C9orf72 FTLD and ALS and reduces poly-GR phase separation and toxicity
   Lauren M. Gittings, Steven Boeynaems, Daniel J. Lightwood, Alison Clargo, Sarfaraj Topia, Lisa Nakayama, Claire Troakes, David M. A. Mann, Aaron D. Gitler, Tammaryn Lashley et al. · 2019 · Acta Neuropathologica
4. ZNF598 co-translationally titrates poly(GR) protein implicated in the pathogenesis of C9ORF72 -associated ALS/FTD
   Jumin Park, Jongbo Lee, Ji-hyung Kim, Jongbin Lee, Heeju Park, Chunghun Lim · 2021 · Nucleic Acids Research
5. Development of Therapeutics for C9ORF72 ALS/FTD-Related Disorders
   Maria Sara Cipolat Mis, Simona Brajkovic, Francesco Tafuri, Nereo Bresolin, Giacomo P. Comi, Stefania Paola Corti · 2016 · Molecular Neurobiology
6. Arginine in C9ORF72 Dipolypeptides Mediates Promiscuous Proteome Binding and Multiple Modes of Toxicity
   Mona Radwan, Ching‐Seng Ang, Angelique R. Ormsby, Dezerae Cox, James C. Daly, Gavin E. Reid, Danny M. Hatters · 2020 · Molecular & Cellular Proteomics
7. Amyotrophic Lateral Sclerosis Genes in Drosophila melanogaster
   Sophie Layalle, Laetitia They, Sarah Ourghani, Cédric Raoul, Laurent Soustelle · 2021 · International Journal of Molecular Sciences
8. Where and Why Modeling Amyotrophic Lateral Sclerosis
   Francesco Liguori, Susanna Amadio, Cinzia Volonté · 2021 · International Journal of Molecular Sciences
9. The regulation of the small heat shock protein B8 in misfolding protein diseases causing motoneuronal and muscle cell death
   Cristofani, Riccardo, Rusmini, Paola, Galbiati, Mariarita, Cicardi, Maria Elena, Ferrari, Veronica, Tedesco, Barbara, Casarotto, Elena, Chierichetti, Marta, Messi, Elio, Piccolella, Margherita et al. · 2019 · Frontiers in Neuroscience
10. Drosophila melanogaster As a Model Organism to Study RNA Toxicity of Repeat Expansion-Associated Neurodegenerative and Neuromuscular Diseases
    Alex Chun Koon, Ho Yin Edwin Chan · 2017 · Frontiers in Cellular Neuroscience
11. Poly-PR in C9ORF72-Related Amyotrophic Lateral Sclerosis/Frontotemporal Dementia Causes Neurotoxicity by Clathrin-Dependent Endocytosis
    Rui Wang, Xingyun Xu, Zongbing Hao, Shun Zhang, Dan Wu, Hongyang Sun, Chenchen Mu, Haigang Ren, Guanghui Wang · 2019 · Neuroscience Bulletin
12. Heterogeneous ribonuclear protein A3 (hnRNP A3) is present in dipeptide repeat protein containing inclusions in Frontotemporal Lobar Degeneration and Motor Neurone disease associated with expansions in C9orf72 gene
    Yvonne S. Davidson, Louis Flood, Andrew Robinson, Yoshihiro Nihei, Kohji Mori, Sara J. Rollinson, Anna M. T. Richardson, Bridget C. Benson, Matthew S. Jones, Julie S. Snowden et al. · 2017 · Acta Neuropathologica Communications
13. Arginine-rich Peptides Can Actively Mediate Liquid-liquid Phase Separation
    Steven Boeynaems, Mathias De Decker, Péter Tompa, Ludo Van Den Bosch · 2017 · BIO-PROTOCOL
14. Nucleolar stress in C9orf72 and sporadic ALS spinal motor neurons precedes TDP-43 mislocalization
    Olubankole Aladesuyi Arogundade, Sandra Nguyen, Ringo Leung, Danielle Wainio, Maria José Gómez-Rodríguez, John Ravits · 2021 · Acta Neuropathologica Communications
15. Fly for ALS: Drosophila modeling on the route to amyotrophic lateral sclerosis modifiers
    Francesco Liguori, Susanna Amadio, Cinzia Volonté · 2021 · Cellular and Molecular Life Sciences
16. Co-expression of C9orf72 related dipeptide-repeats over 1000 repeat units reveals age- and combination-specific phenotypic profiles in Drosophila
    Ryan J. H. West, Joanne L. Sharpe, André Voelzmann, Anna L. Munro, Ines Hahn, Richard Baines, Stuart M. Pickering-Brown · 2020 · Acta Neuropathologica Communications
17. Mitochondria Dysfunction in Frontotemporal Dementia/Amyotrophic Lateral Sclerosis: Lessons From Drosophila Models
    Sharifah Anoar, Nathaniel S. Woodling, Teresa Niccoli · 2021 · Frontiers in Neuroscience
18. Nuclear-import receptors as gatekeepers of pathological phase transitions in ALS/FTD
    Bilal Khalil, Miriam Linsenmeier, Courtney L. Smith, James Shorter, Wilfried Rossoll · 2024 · Molecular Neurodegeneration
19. Multiple pathways of toxicity induced by C9orf72 dipeptide repeat aggregates and G4C2 RNA in a cellular model
    Frédéric Frottin, Manuela Pérez‐Berlanga, F. Ulrich Hartl, Mark S. Hipp · 2021 · eLife
20. Molecular basis of C9orf72 poly-PR interference with the β-karyopherin family of nuclear transport receptors
    Hamidreza Jafarinia, Erik Van der Giessen, Patrick R. Onck · 2022 · Scientific Reports
21. Divergence, Convergence, and Therapeutic Implications: A Cell Biology Perspective of C9ORF72-ALS/FTD
    Xiaoqiang Tang, Arturo Toro, T. G. Sahana, Junli Gao, Jessica L Chalk, Björn Oskarsson, Ke Zhang · 2020 · Molecular Neurodegeneration
22. The coming-of-age of nucleocytoplasmic transport in motor neuron disease and neurodegeneration
    Paulo A. Ferreira · 2019 · Cellular and Molecular Life Sciences
23. Poor old pores—The challenge of making and maintaining nuclear pore complexes in aging
    Irina L. Rempel, Anton Steen, Liesbeth M. Veenhoff · 2020 · FEBS Journal
24. CRISPR/Cas9 screen in human iPSC‐derived cortical neurons identifies NEK6 as a novel disease modifier of C9orf72 poly(PR) toxicity
    Wenting Guo, Haibo Wang, Arun Kumar Tharkeshwar, Julien Couthouis, Elke Braems, Pegah Masrori, Evelien Van Schoor, Yannan Fan, Karan Ahuja, Matthieu Moisse et al. · 2022 · Alzheimer s & Dementia
25. The Hitchhiker’s Guide to Nucleocytoplasmic Trafficking in Neurodegeneration
    Stephen R. Moore, Benjamin E. Rabichow, Rita Sattler · 2020 · Neurochemical Research
26. Glial Cell Dysfunction in C9orf72-Related Amyotrophic Lateral Sclerosis and Frontotemporal Dementia
    Mehdi Ghasemi, Kiandokht Keyhanian, Catherine L. Douthwright · 2021 · Cells
27. Altered Phase Separation and Cellular Impact in C9orf72-Linked ALS/FTD
    Daniel A. Solomon, Rebekah Smikle, Matthew J. Reid, Sarah Mizielinska · 2021 · Frontiers in Cellular Neuroscience
28. Novel genetic variants in MAPT and alterations in tau phosphorylation in amyotrophic lateral sclerosis post‐mortem motor cortex and cerebrospinal fluid
    Tiziana Petrozziello, Ana Claudia Silva Amaral, Simon Dujardin, Sali M.K. Farhan, James Gin Man Chan, Bianca A. Trombetta, Pia Kivisäkk, Alexandra N. Mills, Evan A. Bordt, Spencer E. Kim et al. · 2021 · Brain Pathology
29. The Role of Nucleocytoplasmic Transport Defects in Amyotrophic Lateral Sclerosis
    Joni Vanneste, Ludo Van Den Bosch · 2021 · International Journal of Molecular Sciences
30. C9orf72 ALS/FTD dipeptide repeat protein levels are reduced by small molecules that inhibit PKA or enhance protein degradation
    Nausicaa Valentina Licata, Riccardo Cristofani, Sally E. Salomonsson, Katherine M Wilson, Liam Kempthorne, Deniz Vaizoglu, Vito D’Agostino, Daniele Pollini, Rosa Loffredo, Michael Pancher et al. · 2021 · The EMBO Journal
31. Mechanisms Associated with TDP-43 Neurotoxicity in ALS/FTLD
    Marc Shenouda, Ashley B. Zhang, Anna Weichert, Janice Robertson · 2018 · Advances in neurobiology
32. RNA-Binding Proteins and the Complex Pathophysiology of ALS
    Wanil Kim, Do‐Yeon Kim, Kyung‐Ha Lee · 2021 · International Journal of Molecular Sciences
33. Loss of C9orf72 perturbs the Ran-GTPase gradient and nucleocytoplasmic transport, generating compositionally diverse Importin β-1 granules
    Philip McGoldrick, Agnes Lau, Zhipeng You, Thomas M. Durcan, Janice Robertson · 2023 · Cell Reports
34. Aberrant Phase Separation is a Common Killing Strategy of Positively Charged Peptides in Biology and Human Disease
    Steven Boeynaems, X. Rosa Ma, Vivian Yeong, Garrett M. Ginell, Jian-Hua Chen, Jacob A. Blum, Lisa Nakayama, Anushka Sanyal, Adam Briner, Delphi Van Haver et al. · 2023 · bioRxiv (Cold Spring Harbor Laboratory)
35. Yeast screen for modifiers of C9orf72 poly(glycine-arginine) dipeptide repeat toxicity
    Noori Chai, Aaron D. Gitler · 2018 · FEMS Yeast Research
36. Microglial activation in an amyotrophic lateral sclerosis-like model caused by Ranbp2 loss and nucleocytoplasmic transport impairment in retinal ganglion neurons
    Kyoungin Cho, Dosuk Yoon, Minzhong Yu, Neal S. Peachey, Paulo A. Ferreira · 2019 · Cellular and Molecular Life Sciences
37. Studying ALS: Current Approaches, Effect on Potential Treatment Strategy
    Elizaveta Ustyantseva, Sergey P. Medvedev, Suren M. Zakian · 2020 · Advances in experimental medicine and biology
38. Mechanisms of repeat-associated non-AUG translation in neurological microsatellite expansion disorders
    Lydia M. Castelli, Wan-Ping Huang, Ya-Hui Lin, Kung‐Yao Chang, Guillaume M. Hautbergue · 2021 · Biochemical Society Transactions
39. Activation of the Keap1/Nrf2 pathway suppresses mitochondrial dysfunction, oxidative stress, and motor phenotypes in C9orf72 ALS/FTD models
    Wing Hei Au, Leonor Miller‐Fleming, Álvaro Sánchez-Martínez, James Lee, Madeleine J. Twyning, Hiran A. Prag, Laura Raik, Scott P. Allen, Pamela J. Shaw, Laura Ferraiuolo et al. · 2024 · Life Science Alliance
40. Antisense, but not sense, repeat expanded RNAs activate PKR/eIF2α-dependent ISR in C9ORF72 FTD/ALS
    Janani Parameswaran, Nancy Zhang, Elke Braems, Kedamawit Tilahun, Devesh C. Pant, Keena Yin, Seneshaw Asress, Kara Heeren, Anwesha Banerjee, Emma Davis et al. · 2023 · eLife
41. Moderate intrinsic phenotypic alterations in C9orf72 ALS/FTD iPSC-microglia despite the presence of C9orf72 pathological features
    Ileana Lorenzini, Eric B. Alsop, Jennifer Levy, Lauren M. Gittings, Deepti Lall, Benjamin E. Rabichow, Stephen R. Moore, Ryan S. Pevey, Lynette M. Bustos, Camelia Burciu et al. · 2023 · Frontiers in Cellular Neuroscience
42. Heterogeneous ribonuclear protein E2 (hnRNP E2) is associated with TDP-43-immunoreactive neurites in Semantic Dementia but not with other TDP-43 pathological subtypes of Frontotemporal Lobar Degeneration
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43. The ataxin-1 interactome reveals direct connection with multiple disrupted nuclear transport pathways
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44. Nuclear-Import Receptors Counter Deleterious Phase Transitions in Neurodegenerative Disease
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45. Meta-analysis of Genetic Modifiers Reveals Candidate Dysregulated Pathways in Amyotrophic Lateral Sclerosis
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46. C9ORF72 protein function and immune dysregulation in amyotrophic lateral sclerosis
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47. LSM12-EPAC1 defines a neuroprotective pathway that sustains the nucleocytoplasmic RAN gradient
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48. Unraveling the impact of disrupted nucleocytoplasmic transport systems in C9orf72-associated ALS
    Philip McGoldrick, Janice Robertson · 2023 · Frontiers in Cellular Neuroscience
49. RNA Nucleocytoplasmic Transport Defects in Neurodegenerative Diseases
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50. Nucleoporins are degraded via upregulation of ESCRT-III/Vps4 complex in Drosophila models of C9-ALS/FTD
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51. C9ORF72 hexanucleotide repeat expansion: From ALS and FTD to a broader pathogenic role?
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52. Capturing single-molecule properties does not ensure accurate prediction of biomolecular phase diagrams
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53. Repeat-associated non-AUG (RAN) translation mechanisms are running into focus for GGGGCC-repeat associated ALS/FTD
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54. Partners in crime: Proteins implicated in RNA repeat expansion diseases
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55. Drosophila models of amyotrophic lateral sclerosis with defects in RNA metabolism
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56. Non-coding RNA in C9orf72-related amyotrophic lateral sclerosis and frontotemporal dementia: A perfect storm of dysfunction
    Andrew G. L. Douglas · 2018 · Non-coding RNA Research
57. p53 Transactivation Domain Mediates Binding and Phase Separation with Poly-PR/GR
    Sinem Usluer, Emil Spreitzer, Benjamin M. R. Bourgeois, Tobias Madl · 2021 · International Journal of Molecular Sciences
58. C9orf72 polyPR directly binds to various nuclear transport components
    Jafarinia, Hamidreza, van der Giessen, Erik, Onck, Patrick R · 2024 · eLife
59. Energy Homeostasis and Abnormal RNA Metabolism in Amyotrophic Lateral Sclerosis
    Yu‐Ju Liu, Po-Yi Tsai, Yijuang Chern · 2017 · Frontiers in Cellular Neuroscience
60. Drosophila as a Model for Assessing the Function of RNA-Binding Proteins during Neurogenesis and Neurological Disease
    Eugenia C. Olesnicky, Ethan G. Wright · 2018 · Journal of Developmental Biology
61. A Chemical Screen Identifies Compounds Limiting the Toxicity of C9ORF72 Dipeptide Repeats
    Alba Corman, Bomi Jung, Maria Häggblad, Lars Bräutigam, Vanesa Lafarga, Louise Lidemalm, Daniela Hühn, Jordi Carreras Puigvert, Óscar Fernández-Capetillo · 2018 · Cell chemical biology
62. Simple animal models for amyotrophic lateral sclerosis drug discovery
    Shunmoogum A. Patten, J. Alex Parker, Xiao‐Yan Wen, Pierre Drapeau · 2016 · Expert Opinion on Drug Discovery
63. A behavioral screen for mediators of age-dependent TDP-43 neurodegeneration identifies SF2/SRSF1 among a group of potent suppressors in both neurons and glia
    Jorge Azpurua, Enas A. Elkarim, Marvel Tranquille, Josh Dubnau · 2021 · PLoS Genetics
64. Inducible expression of human C9ORF72 36× G4C2 hexanucleotide repeats is sufficient to cause RAN translation and rapid muscular atrophy in mice
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65. Exploring the alternative: Fish, flies and worms as preclinical models for ALS
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66. Phenotypic Suppression of ALS/FTD-Associated Neurodegeneration Highlights Mechanisms of Dysfunction
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67. Measuring and Interpreting Nuclear Transport in Neurodegenerative Disease—The Example of C9orf72 ALS
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68. C9orf72-linked arginine-rich dipeptide repeats aggravate pathological phase separation of G3BP1
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69. Review: Modelling the pathology and behaviour of frontotemporal dementia
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70. Regulating Phase Transition in Neurodegenerative Diseases by Nuclear Import Receptors
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71. Zfp106 binds to G-quadruplex RNAs and inhibits RAN translation and formation of RNA foci caused by G4C2 repeats
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72. Hypothesis and Theory: Roles of Arginine Methylation in C9orf72-Mediated ALS and FTD
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73. Epigenetic Small Molecules Rescue Nucleocytoplasmic Transport and DNA Damage Phenotypes in C9ORF72 ALS/FTD
    Melina Ramic, Nadja S. Andrade, Matthew J. Rybin, Rustam Esanov, Claes Wahlestedt, Michael G Benatar, Zane R. Zeier · 2021 · Brain Sciences
74. Repeat Expansion Disease Models
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75. Artificial microRNA suppresses C9ORF72 variants and decreases toxic dipeptide repeat proteins in vivo
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76. The nuclear ubiquitin ligase adaptor SPOP is a conserved regulator of C9orf72 dipeptide toxicity
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77. Parallel import mechanisms ensure the robust nuclear localization of actin in Drosophila
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78. Knockdown of genes involved in axonal transport enhances the toxicity of human neuromuscular disease‐linked MATR3 mutations in Drosophila
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79. Insights into disease mechanisms and potential therapeutics for C9orf72-related amyotrophic lateral sclerosis/frontotemporal dementia
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80. Modeling C9orf72-Related Frontotemporal Dementia and Amyotrophic Lateral Sclerosis in Drosophila
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81. C9orf72 -associated arginine-rich dipeptide repeats induce RNA-dependent nuclear accumulation of Staufen in neurons
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82. Cellular Stress Induces Nucleocytoplasmic Transport Deficits Independent of Stress Granules
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83. Proline‐arginine poly‐dipeptide encoded by the C9orf72 repeat expansion inhibits adenosine deaminase acting on RNA
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84. Proteostasis deregulation as a driver of C9ORF72 pathogenesis
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85. Brain Cell Type-Specific Nuclear Proteomics Is Imperative to Resolve Neurodegenerative Disease Mechanisms
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86. Models and mechanisms of repeat expansion disorders: a worm’s eye view
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87. Nuclear transport under stress phenocopies transport defects in models of C9Orf72 ALS
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88. The nuclear import receptor Kapβ2 modifies neurotoxicity mediated by poly(GR) in C9orf72-linked ALS/FTD
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89. A novel overlapping NLS/NES region within the PH domain of Rho Guanine Nucleotide Exchange Factor (RGNEF) regulates its nuclear-cytoplasmic localization
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90. Toxicity of C9orf72 -associated dipeptide repeat peptides is modified by commonly used protein tags
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91. Viruses in Laboratory Drosophila and Their Impact on Host Gene Expression
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92. Nucleus–cytoplasm cross‐talk in the aging brain
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93. Transcriptome-wide RNA binding analysis of C9orf72 poly(PR) dipeptides
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94. Neural stem cells alter nucleocytoplasmic partitioning and accumulate nuclear polyadenylated transcripts during quiescence
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95. Altered nuclear envelope homeostasis is a key pathogenic event in C9ORF72-linked ALS/FTD
    Riccardo Sirtori, Michelle Gregoire, Alicia Collins, Serena Santangelo, Bhavya Chatragadda, Robert Cullen, Antonia Ratti, Claudia Fallini · 2024 · bioRxiv (Cold Spring Harbor Laboratory)
96. NLS-binding deficient Kapβ2 reduces neurotoxicity via selective interaction with C9orf72-ALS/FTD dipeptide repeats
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97. The Nuclear Envelope in Ageing and Progeria
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98. Aspects of degradation and translation of the expanded C9orf72 hexanucleotide repeat RNA
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99. Modeling Myotonic Dystrophy Type 2 Using Drosophila melanogaster
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100. A comparison study of pathological features and drug efficacy between Drosophila models of C9orf72 ALS/FTD
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